Research into Spinal Deformities 6

RESEARCH INTO SPINAL DEFORMITIES 6 Studies in Health Technology and Informatics This book series was started in 1990 t...

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RESEARCH INTO SPINAL DEFORMITIES 6

Studies in Health Technology and Informatics This book series was started in 1990 to promote research conducted under the auspices of the EC programmes’ Advanced Informatics in Medicine (AIM) and Biomedical and Health Research (BHR) bioengineering branch. A driving aspect of international health informatics is that telecommunication technology, rehabilitative technology, intelligent home technology and many other components are moving together and form one integrated world of information and communication media. The complete series has been accepted in Medline. Volumes from 2005 onwards are available online. Series Editors: Dr. J.P. Christensen, Prof. G. de Moor, Prof. A. Famili, Prof. A. Hasman, Prof. L. Hunter, Dr. I. Iakovidis, Dr. Z. Kolitsi, Mr. O. Le Dour, Dr. A. Lymberis, Prof. P.F. Niederer, Prof. A. Pedotti, Prof. O. Rienhoff, Prof. F.H. Roger France, Dr. N. Rossing, Prof. N. Saranummi, Dr. E.R. Siegel, Dr. P. Wilson, Prof. E.J.S. Hovenga, Prof. M.A. Musen and Prof. J. Mantas

Volume 140 Recently published in this series Vol. 139. A. ten Teije, S. Miksch and P. Lucas (Eds.), Computer-based Medical Guidelines and Protocols: A Primer and Current Trends Vol. 138. T. Solomonides et al. (Eds.), Global Healthgrid: e-Science Meets Biomedical Informatics – Proceedings of HealthGrid 2008 Vol. 137. L. Bos, B. Blobel, A. Marsh and D. Carroll (Eds.), Medical and Care Compunetics 5 Vol. 136. S.K. Andersen, G.O. Klein, S. Schulz, J. Aarts and M.C. Mazzoleni (Eds.), eHealth Beyond the Horizon – Get IT There – Proceedings of MIE2008 – The XXIst International Congress of the European Federation for Medical Informatics Vol. 135. T.B. Grivas (Ed.), The Conservative Scoliosis Treatment – 1st SOSORT Instructional Course Lectures Book Vol. 134. B. Blobel, P. Pharow and M. Nerlich (Eds.), eHealth: Combining Health Telematics, Telemedicine, Biomedical Engineering and Bioinformatics to the Edge – Global Experts Summit Textbook Vol. 133. J. Hammer, M. Nerlich and S. Dendorfer (Eds.), Medicine Meets Engineering – Proceedings of the 2nd Conference on Applied Biomechanics Regensburg Vol. 132. J.D. Westwood, R.S. Haluck, H.M. Hoffman, G.T. Mogel, R. Phillips, R.A. Robb and K.G. Vosburgh (Eds.), Medicine Meets Virtual Reality 16 – parallel, combinatorial, convergent: NextMed by Design Vol. 131. R. Latifi (Ed.), Current Principles and Practices of Telemedicine and e-Health

ISSN 0926-9630

Research into Spinal Deformities 6

Edited by

Peter H. Dangerfield The University of Liverpool, Royal Liverpool Children’s Hospital, Liverpool and Staffordshire University, Stoke on Trent, UK

Amsterdam • Berlin • Oxford • Tokyo • Washington, DC

© 2008 The authors and IOS Press. All rights reserved. No part of this book may be reproduced, stored in a retrieval system, or transmitted, in any form or by any means, without prior written permission from the publisher. ISBN 978-1-58603-888-5 Library of Congress Control Number: 2008929935 Publisher IOS Press Nieuwe Hemweg 6B 1013 BG Amsterdam Netherlands fax: +31 20 687 0019 e-mail: [email protected] Distributor in the UK and Ireland Gazelle Books Services Ltd. White Cross Mills Hightown Lancaster LA1 4XS United Kingdom fax: +44 1524 63232 e-mail: [email protected]

Distributor in the USA and Canada IOS Press, Inc. 4502 Rachael Manor Drive Fairfax, VA 22032 USA fax: +1 703 323 3668 e-mail: [email protected]

LEGAL NOTICE The publisher is not responsible for the use which might be made of the following information. PRINTED IN THE NETHERLANDS

Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved.

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Preface The International Research Society for Spinal Deformities (IRSSD) was founded in 1994. The first concepts of forming the Society were raised by a group of researchers who had first gathered in Vermont in 1980 to hold what subsequently became a series of biennial meetings devoted to surface topography and deformity. At a meeting held in 1992 in Montreal, focussed on 3 dimensional interpretations of spinal deformities, discussions were held into whether a formal Society should be formed, and while debate rejected this concept at the time, it was two years later at a meeting in Pescara, Italy 1994 that the IRSSD finally came into being. The Society has its roots in mathematical modelling, biomechanics and medical research and has always encouraged researchers to report their on-going projects in all aspects of the spinal deformities associated with scoliosis and other spinal deformity conditions. The biomechanical approach led to a better understanding of how the vertebral column behaves under stresses and how the surface shape related to the underlying columnar deformity. Developments in imaging and electronics allowed researchers to develop systems such as ISIS as a non-invasive method of recording back shape in out patient clinics as well as a tool for researchers. Biologically based studies were also reported. It was appreciated early on by the membership that children with scoliosis have a range of growth disturbances associated with a marked skeletal asymmetry. While the biological approach has enabled a considerable amount of data to be amassed which relates to etiology and pathogenesis, no actual breakthrough into fully understanding why the spinal column rotates and curves to cause scoliosis has yet occurred. The biennial meetings of the Society chart this research progress in each of the volumes of proceedings, with some reports being final papers while others are progress reports. This is a unique resource for the researcher, holding the key to many different aspects of the problems of the spine. However, some authors choose not to submit to the volumes as they are publishing elsewhere in peer-reviewed Journals, a trend which is inevitably going to challenge the way conference proceedings are reported in the future. There is therefore a challenge to the IRSSD to find an answer to this issue. 2008 could be viewed as a time of potentially exciting breakthroughs in our understanding of the deformity of scoliosis. The rapid advances in imaging technology will allow better and more detailed images of the spine, both on its surface and deeper internally, using techniques such as Laser scanning, Magnetic Resonance Imaging and ultrasound. There is little doubt these technologies are going to advance massively, new ones will come into the hands of clinicians and researchers and a better understanding of the complex functional anatomy of the spine will be developed. This will undoubtedly aid biomechanicians to model the spine and its function, under gravity and movement, allowing new insight into progressions of curves and ways to surgically control deformities. However, it is probably the potential of biology and medical research which offer the greatest opportunities to further our understanding. The incredible advances at the molecular level, the expanding knowledge of genetics and the recent discoveries in the field of neurophysiology offer for the first time real potential for unravelling the puz-

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zles of etiology. The discovery of molecular biochemical pathways, signalling agents, hormones such as leptin, genetic markers and a greater appreciation of the role of the nervous systems, both central, peripheral and autonomic, all indicate that the research field could expand dramatically with new ideas and inputs from research workers in fields outside the traditional ones devoted to the spine. It is thus clear that to find a solution, we must build on these new developments and look beyond the spine to the whole body to discover if its biological functions are disturbed. A growing child is a dynamic environment internally, with rapid growth changes reflected in adolescence and these are known to result in tall, thin and asymmetrical children. These changes point to a whole body biological involvement. Researchers must reflect on this totality since it must ultimately allow us to explain the etiology and pathology of what are probably going to prove to be secondary events manifest as spinal curvatures. These are exciting times and I hope the participants in the Liverpool meeting in 2008 will look back on it as a watershed in our understanding of the spinal deformities. Let’s hope this is true, since a therapeutic intervention must surely be better than surgery or external bracing from the purely psychological, if not cosmetic, view of a growing child. I would like to thanks all participants and authors for submitting their work to the meeting and to my colleagues Professor Nachi Chockalingam, Mr. Ashley Cole and Mr. El-Nasri Ahmed, for their help and support and to our sponsors who supported the meeting in Liverpool, European Capital of Culture 2008. Peter Dangerfield Liverpool, UK April 2008

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Contents Preface Peter Dangerfield

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Chapter 1. Genetics Cytogenic Abnormalities in Buccal Cells During Spinal Deformities in Children M.Y. Karganov, M.I. Kozhevnikva, A.V. Aleschenko, N.N. Khlebnikova, I.B. Alchinova, I.I. Pelevina, A.M. Serebranyi, K.S. Ternovoy and L.A. Noskin

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Chapter 2. Growth Body Mass Index of Girls in Health Influences Menarche and Skeletal Maturation: A Leptin-Sympathetic Nervous System Focus on the Trunk with Hypothalamic Asymmetric Dysfunction in the Pathogenesis of Adolescent Idiopathic Scoliosis? R.G. Burwell, R.K. Aujla, A.S. Kirby, P.H. Dangerfield, A. Moulton, A.A. Cole, F.J. Polak, R.K. Pratt and J.K. Webb Volumetric Thoracic Growth in Children with Moderate and Severe Scoliosis Compared to Subjects Without Spinal Deformity Y.P. Charles, A. Dimeglio, M. Marcoul, J.-F. Bourgin, A. Marcoul and M.-C. Bozonnat Back Trunk Morphology in 3301 Children Aged 3–9 Years Old T.B. Grivas, E.S. Vasiliadis, C. Mihas, C. Maziotou and G. Triandafyllopoulos The Role of the Intervertebral Disc in Correction of Scoliotic Curves. A Theoretical Model of Idiopathic Scoliosis Pathogenesis T.B. Grivas, E.S. Vasiliadis, G. Rodopoulos and N. Bardakos Ultrasound Femoral Anteversion (FAV) Relative to Tibial Torsion (TT) is Abnormal After School Screening for Adolescent Idiopathic Scoliosis (AIS): Evaluation by Two Methods R.G. Burwell, R.K. Aujla, A.S. Kirby, A. Moulton, P.H. Dangerfield, B.J.C. Freeman, A.A. Cole, F.J. Polak, R.K. Pratt and J.K. Webb Risser Sign: The Value of the Lateral Spinal Radiograph to Assess the Excursion of the Iliac Apophysis T. Kotwicki Stature and Growth Compensation for Spinal Curvature I.A.F. Stokes Clinical Detectable Tension in the Growing Body: New and Revisited Signs in Clinical Examination in Children with Postural Problems and Spinal Deformities P.J.M. van Loon

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Chapter 3. Biomechanics, Surface Topography and Imaging Quantification of Intervertebral Efforts During Walking: Comparison Between a Healthy and a Scoliotic Subject M. Raison, C.-É. Aubin, C. Detrembleur, P. Fisette and J.-C. Samin

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Measuring the Rib Hump in Scoliosis with ISIS2 F. Berryman, P. Pynsent and J. Fairbank Thoracic Kyphosis Angle Measurements with ISIS2 F. Berryman, P. Pynsent and J. Fairbank Sagittal Alignment Correction of the Thoracolumbar Junction in Idiopathic Scoliosis by in Situ Bending Technique Y.P. Charles, N. Meyer and J.-P. Steib A 3-D Skeleton Model & SEMG Approach for Integrated Neck and Low Back Pain Analysis Test Batteries M. D’Amico, G. D’Amico, M. Frascarello, M. Paniccia, P. Roncoletta and M. Vallasciani Analysis of the Mechanisms of Idiopathic Scoliosis Progression Using Finite Element Simulation X. Drevelle, J. Dubousset, Y. Lafon, E. Ebermeyer and W. Skalli The Relationship Between Hip Flexion/Extension and the Sagittal Curves of the Spine C. Driscoll, C.-E. Aubin, H. Labelle and J. Dansereau Constrained Intensity-Based Image Registration: Application to Aligning Human Back Images A.S. Elsafi, N.G. Durdle and J.V. Raso Ergonomically Designed Kneeling Chairs Are They Worth It?: Comparison of Sagittal Lumbar Curvature in Two Different Seating Postures J. Bettany-Saltikov, J. Warren and M. Jobson Faulty Posture and Style of Life in Young Adults A. Czakwari, K. Czernicki and J. Durmala Finite Element Modeling of Vertebral Body Stapling Applied for the Correction of Idiopathic Scoliosis: Preliminary Results N.M. Lalonde, C.-É. Aubin, R. Pannetier and I. Villemure Influence of Correction Objectives on the Optimal Scoliosis Instrumentation Strategy: A Preliminary Study Y. Majdouline, C.-É. Aubin and H. Labelle Development of an Apparatus to Evaluate Adolescent Idiopathic Scoliosis by Dynamic Surface Topography T.M.L. Shannon Biomechanical Modelling of a Direct Vertebral Translation Instrumentation System: Preliminary Results X. Wang, C.-É. Aubin, H. Labelle and D. Crandall SpineCor vs. Natural History – Explanation of the Results Obtained Using a Simple Biomechanical Model H.-R. Weiss Comparison of the Kyphosis Angle Evaluated by Video Rasterstereography (VRS) with X-Ray Measurements H.-R. Weiss and N. Elobeidi Postural Changes in Patients with Scoliosis in Different Postural Positions Revealed by Surface Topography K. Schumann, I. Püschel, A. Maier-Hennes and H.-R. Weiss Quantification of Localized Vertebral Deformities Using a Sparse Wavelet-Based Shape Model R. Zewail, A. Elsafi and N. Durdle

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Computer-Assisted Cobb Angle Measurement on Posteroanterior Radiographs J. Zhang, E. Lou, L.H. Le, D. Hill, J. Raso and Y. Wang New Method of Scoliosis Deformity Assessment: ISIS2 System A. Zubović, N. Davies, F. Berryman, P. Pynsent, N. Quraishi, C. Lavy, G. Bowden, J. Wilson-MacDonald and J. Fairbank A Novel Solution for Registration of Stereo Digital Torso Images of Scoliosis Patients A. Kumar, N. Durdle and J. Raso Marker Placement for Movement Analysis in Scoliotic Patients: A Critical Analysis of Existing Systems N. Chockalingam, T.L. Chevalier, M.K. Young and P.H. Dangerfield The Central Cord-Nervous Roots Complex and the Formation and Deformation of the Spine; The Scientific Work on Systematic Body Growth by Milan Roth of Brno (1926–2006) P.J.M. van Loon and L.W. van Rhijn

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Chapter 4. Etiology Leg-Arm Length Ratios Correlate with Severity of Apical Vertebral Rotation in Girls After School Screening for Adolescent Idiopathic Scoliosis (AIS): A Dynamic Pathomechanism in the Initiation of the Deformity? R.G. Burwell, R.K. Aujla, A.S. Kirby, P.H. Dangerfield, A. Moulton, B.J.C. Freeman, A.A. Cole, F.J. Polak, R.K. Pratt and J.K. Webb New Clinical Observations Connected with “Biomechanical Aetiology of so Called Idiopathic Scoliosis” (2006–2007) T. Karski Etiologic Theories of Idiopathic Scoliosis: Autonomic Nervous System and the Leptin-Sympathetic Nervous System Concept for the Pathogenesis of Adolescent Idiopathic Scoliosis R.G. Burwell, P.H. Dangerfield, A. Moulton and S.I. Anderson Etiologic Theories of Idiopathic Scoliosis. Somatic Nervous System and the NOTOM Escalator Concept as One Component in the Pathogenesis of Adolescent Idiopathic Scoliosis R.G. Burwell, P.H. Dangerfield and B.J.C. Freeman

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Chapter 5. Screening and Prevention of Scoliosis Carrying a Rucksack on Either Shoulder or the Back, Does It Matter? Load Induced Functional Scoliosis in “Normal” Young Subjects J. Bettany-Saltikov, J. Warren and M. Stamp Ultrasound Femoral Anteversion (FAV) and Tibial Torsion (TT) After School Screening for Adolescent Idiopathic Scoliosis (AIS) R.G. Burwell, R.K. Aujla, A.S. Kirby, A. Moulton, P.H. Dangerfield, B.J.C. Freeman, A.A. Cole, F.J. Polak, R.K. Pratt and J.K. Webb Positional Relationship Between Leg Rotation and Lumbar Spine During Quiet Standing N. Parker, A. Greenhalgh, N. Chockalingam and P.H. Dangerfield

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Aetiology of Idiopathic Scoliosis. What Have We Learned from School Screening? T.B. Grivas, E.S. Vasiliadis and G. Rodopoulos Suggestions for Improvement of School Screening for Idiopathic Scoliosis T.B. Grivas, E.S. Vasiliadis and J.P. O’Brien Can Future Back Pain in AIS Subjects Be Predicted During Adolescence from the Severity of the Deformity? D. Hill, E. Parent, E. Lou and J. Mahood A Machine Learning Approach to Assess Changes in Scoliosis L. Ramirez, N.G. Durdle and V.J. Raso

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Chapter 6. Mathematical Modelling The Posterior Skeletal Thorax: Rib-Vertebral Angle and Axial Vertebral Rotation Asymmetries in Adolescent Idiopathic Scoliosis 263 R.G. Burwell, R.K. Aujla, B.J.C. Freeman, P.H. Dangerfield, A.A. Cole, A.S. Kirby, F.J. Polak, R.K. Pratt and A. Moulton Prediction of the T2-T12 Kyphosis in Adolescent Idiopathic Scoliosis Using a Multivariate Regression Model 269 S. Kadoury, F. Cheriet and H. Labelle Intervertebral Disc Changes in an Animal Model Representing Altered Mechanics in Scoliosis 273 I.A.F. Stokes, C.A. McBride and D.D. Aronsson Chapter 7. Education Knowledge About Idiopathic Scoliosis Among Students of Physiotherapy D. Ciazynski, K. Czernicki and J. Durmala

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Chapter 8. Treatment of Scoliosis Thoracoplasty in the Surgical Treatment of Adolescent Idiopathic Scoliosis T. Greggi, G. Bakaloudis, F. Lolli, M. Di Silvestre, A. Cioni, S. Giacomini, G. Barbanti Brodano, F. Vommaro, K. Martikos and P. Parisini Preliminary Validation of Curve Progression Model for Brace Treatment E. Lou, D. Hill, E. Parent, J. Raso, J. Mahood, M. Moreau and D. Hedden BRACE MAP, a Proposal for a New Classification of Braces S. Negrini, F. Zaina and S. Atanasio Clinical and Postural Behaviour of Scoliosis During Daily Brace Weaning Hours S. Negrini, C. Fusco, M. Romano, F. Zaina and S. Atanasio Do Imbalance Situations Stimulate a Spinal Straightening Reflex in Patient with Adolescent Idiopathic Scoliosis? M. Romano, V. Ziliani, S. Atanasio, F. Zaina and S. Negrini Congenital Scoliosis – Presentation of Three Severe Cases Treated Conservatively H.-R. Weiss Conservative Scoliosis Treatment in Patients with Prader-Willi Syndrome H.-R. Weiss and S. Bohr

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Chapter 9. Abstracts FBN3 Gene Polymorphisms in Adolescent Idiopathic Scoliosis Patients Xing-bing Cao, Yong Qiu, Xu-sheng Qiu, Zhi-jun Chen, Hai-ou Chen and Wen-jun Chen Investigation on the Association Between Estrogen β Receptor Gene Polymorphisms and the Susceptibility of Adolescent Idiopathic Scoliosis Hai-ou Chen, Zhi-jun Chen, Yong Qiu, Xu-sheng Qiu, Xing-bing Cao, Zhen Liu and Wei-guo Li Vitamin D Receptor Gene Polymorphisms: No Association with Low Bone Mineral Density in Adolescent Idiopathic Scoliosis Girls Wen-Jun Chen, Yong Qiu, Xing-Bing Cao, Xu-Sheng Qiu, Zhi-Jun Chen and Hai-Ou Chen Expression and Significance of Sox9 in Chondrocyte Cells from Adolescent Idiopathic Scoliosis Patients A. Huang, Y. Qiu and G. Sun Promoter Polymorphism of Matrilin-1 Gene (MATN1) Is Associated with Susceptibility to Adolescent Idiopathic Scoliosis: A Case-Control Study Y. Qiu, Z. Chen, N.L.-S. Tang and J.C.-Y. Cheng Left-Right Asymmetry Gene Expression Domains Are Reversed in Adolescent Idiopathic Scoliosis A. Moreau, B. Azeddine, D.S. Wang, H. Labelle, B. Poitras, C.-H. Rivard, G. Grimard, J. Ouellet and S. Parent SNPs Analysis of CHD7 Gene and Idiopathic Scoliosis (IS) in Chinese N.L. Tang, H.Y. Yeung, H. Fan, R. Kwok, V.W. Hung, K.M. Lee, T.P. Lam, B.W. Ng, Y. Qiu and J.C. Cheng Collagen I Alpha 2 Gene Polymorphism Association Study in Adolescent Idiopathic Scoliosis H.Y. Yeung, N.L. Tang, V.W.Y. Hung, R. Kwok, K.M. Lee, L. Qin, B.K.W. Ng and J.C.Y. Cheng A Genetic Model for Studying the Etiopathogenesis of Late-Onset Idiopathic Scoliosis N.L.S. Tang, H.Y. Yeung, K.M. Lee, X. Qiu, Y. Qiu and J.C.Y. Cheng Physical Exercises and Adolescent Idiopathic Scoliosis: Results of a Comprehensive Systematic Review of the Literature M. Romano, C. Fusco, S. Minozzi, S. Atanasio, F. Zaina and S. Negrini Identification of SRS 22r Domains Using Factor Analysis Methodology D.C. Burton, S.M. Lai and M.A. Asher Variations of Semicircular Canals Orientation and Left-Right Asymmetry in Adolescent Idiopathic Scoliosis (AIS) Comparing with Normal Controls: MR Morphometry Study Using Advanced Image Computation Techniques W.C.W. Chu, L. Shi, D. Wang, T. Paus, R.G. Burwell, G.C.W. Man, A. Cheng, H.Y. Yeung, K.M. Lee, P.A. Heng and J.C.Y. Cheng The SRS Outcome Questionnaire Can Discriminate Between Patients with Spondylolisthesis and Normal Healthy Adolescents S. Parent, J. Joncas, M. Roy-Beaudry, M. Beausejour, G. Grimard, M. Forcier and H. Labelle

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The Use of a Decision Tree Increases Accuracy when Classifying Adolescent Idiopathic Scoliosis Using Lenke Classification P. Phan, N. Mezghani, S. Parent, J.A. de Guise and H. Labelle Are There Sub-Types in Lenke-1 Curves? A.P. Sangole, C.-E. Aubin, I.A.S. Stokes and H. Labelle Posterior Instrumented Scoliosis Correction in Thoracic Late Onset Idiopathic Scoliosis: Two Year Results A.A. Cole, S.A. Qaimkhani, S. Sharma, B. Naylor, C. Hughes, L.M. Breakwell and D.L. Douglas Can Surgical Reduction Correct Spino-Pelvic Alignment in L5-S1 Developmental Spondylolisthesis? H. Labelle, P. Roussouly, D. Chopin, E. Berthonnaud, T. Hresko and M. O’Brien The Relationship Between Pelvic Balance and a Dome-Shaped Sacrum in Developmental Spondylolisthesis H. Labelle, P. Roussouly, É. Berthonnaud, S. Hu and C. Brown A Historical and Observational Study to Expose Some Etiopathogenetic Factors that May Be Relevant to Adolescent Idiopathic Scoliosis (AIS) in Some Patients M.E. McMaster French-Canadian Validation of the Spinal Appearance Questionnaire (SAQ) in the Adolescent Patients at a Scoliosis Clinic and Its Clinical Application S. Parent, M. Roy-Beaudry, J. Joncas, M. Beauséjour, M. Forcier, S. Bekhiche, G. Grimard and H. Labelle An Energy-Based Mechanobiological Growth Model for Simulating Vertebral Progressive Deformities Under Multi-Direction Loads H. Lin, C.-É. Aubin, I. Villemure and S. Parent US and European Risser Grading Systems: Which One Best Predict the Curve Acceleration Phase in Adolescent Idiopathic Scoliosis? M.L. Nault, S. Parent, H. Labelle, M. Roy-Beaudry and M. Rivard Decreased Lean Mass in Adolescent Idiopathic Scoliosis Weiguo Li and Yong Qiu The Relationship Between RANKL/OPG and the Decreased Bone Mass in Adolescent Idiopathic Scoliosis Patients Z. Liu, Y. Qiu, B. Wang, W. Ma, F. Zhu, Z. Zhu, Y. Yu and B. Qian Association Between Growth Hormone Gene and Adolescent Idiopathic Scoliosis Xu-Sheng Qiu, Nelson Leung-Sang Tang, Hiu-Yen Yeung, Jack Chun-Yiu Cheng and Yong Qiu Expression of Melatonin Receptor in Chondrocyte of Adolescent Idiopathic Scoliosis Guangquan Sun, Weijun Wang, Aibing Huang, Jack Chun-Yiu Cheng and Yong Qiu mRNA Expressions of Type I Collagen, Osteocalcin and Osteoprotegerin in Iliac Cancellous Bone from Girls with Adolescent Idiopathic Scoliosis Xu Sun, Yong Qiu, Weiguo Li, Bin Wang, Weiwei Ma, Feng Zhu, Zezhang Zhu, Yang Yu and Bangping Qian Expression of Runx2 and Type X Collagen in Human Vertebral Growth Plate of Adolescent Idiopathic Scoliosis Shoufeng Wang, Yong Qiu, Weiguo Li, Bin Wang, Weiwei Ma, Feng Zhu, Zezhang Zhu, Yang Yu and Bangping Qian

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Is Corticospinal Tract Organization Different in Idiopathic Scoliosis? D. Mihaila and B. Calancie Effects of in vivo Mechanical Loading on Extracellular Matrix Components of the Growth Plate M. Cancel, I. Villemure, F. Moldovan and G. Grimard Transverse Plane Pelvic Rotation Following Rotationally Corrective Instrumentation of Adolescent Idiopathic Scoliosis Double Curves M.A. Asher, D.C. Burton, S.M. Lai, J.L. Gum and B. Carlson A Multivariate Regression Model for Predicting the T2-T12 Kyphosis in Adolescent Idiopathic Scoliosis S. Kadoury, F. Cheriet and H. Labelle Poor Bone Mechanical and Structural Properties in 528 AIS Patients – Using Non-Invasive Quantitative Ultrasound V.W.Y. Hung, L. Qin, H.Y. Yeung, W.T.K. Lee, K.M. Lee and J.C.Y. Cheng The Effect on the Intervertebral Pressure Distribution in a Goat Spine upon Implementation of a Spring-Like Device X.C. Liu, R. Rizza, J. Thometz, C. Tassone and R. Lyon Stress Shielding Within the Scoliotic Spine: A Progressive Risk Factor? M. Driscoll, C.-É. Aubin, S. Parent and A. Moreau Suspension Is Better than Side-Bending to Assess Spinal Flexibility in Adolescent Idiopathic Scoliosis M.-E. Lamarre, S. Parent, H. Labelle, C.-E. Aubin, J. Joncas and Y. Petit Classification of Scoliosis Deformity 3-D Spinal Shape by Cluster Analysis Ian A.F. Stokes, Archana P. Sangole and Carl-Eric Aubin Alteration of Mobility and Trunk Muscle Activation During Walking and Bending in AIS Patients with Posterior Fusion C. Tassone, X.C. Liu, T. Jones, J. Thometz and R. Lyon Three-Dimensional MRI Analysis of the Skull Morphometry in Adolescent Idiopathic Scoliosis Girls: A Pilot Study H.Y. Yeung, L. Shi, W.C.W. Chu, C.W. Man, T.H. Cheng, V.W.Y. Hung, K.M. Lee, P.A. Heng, B.K.W. Ng and J.C.Y. Cheng Enhancement of Low Intensity Pulsed Ultrasound on Spinal Fusion Augmented with Stem Cell-Bioceramic Composite Chun Wai Chan, Celine F.F. Hui, Wei Man Pan, Kwong Man Lee, Ling Qin, Yun Yu Hu, Kwok Sui Leung and Jack C.Y. Cheng A New System for Classifying Torso Deformities in Scoliosis Using Localized Shape Indices P.O. Ajemba, N.G. Durdle and V.J. Raso A Novel Visualization Scheme for Monitoring and Tracking of Torso Deformities in Scoliosis P.O. Ajemba, N.G. Durdle and V.J. Raso Analysis of Trunk External Asymmetry in Side-Bending Valérie Pazos, Fethia Miled, Philippe Debanné, Hubert Labelle and Farida Cheriet Sagittal Spino-Pelvic Alignment in Lumbosacral Spondylolisthesis Jean-Marc Mac-Thiong, Zhi Wang, Jacques A. de Guise and Hubert Labelle

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Cerebrospinal Fluid (CSF) Flow Dynamics at the Craniocervical Junction in Adolescent Idiopathic Scoliosis: Morphological and Functional Study with Phase Contrast Magnetic Resonance Imaging W.C.W. Chu, G.C.W. Man, W.W.M. Lam, H.Y. Yeung, B.K.W. Ng, T.P. Lam, K.M. Lee and J.C.Y. Cheng Is There Any Regional Difference of Brain Tissue Densities Between Adolescent Idiopathic Scoliosis (AIS) Patients and Normal Controls: A Morphometric Study with High Resolution MR Brain Imaging W.C.W. Chu, L. Shi, D. Wang, T. Paus, A. Pitiot, R.G. Burwell, G.C.W. Man, A. Cheng, H.Y. Yeung, K.M. Lee, P.A. Heng, B. Freeman and J.C.Y. Cheng A Significant Number of Patients with AIS Exhibit Transverse Plane Pelvis Deformation J.E. Meunier, C.-E. Aubin, H. Labelle, A. Sangole, R. Jackson, L. Lenke and P. Newton Reducing Radiation Exposure for Scoliosis S. Parent, S. Deschênes, G. Charron, G. Beaudoin, M.-C. Miron, J. Dubois and H. Labelle Abnormal Proliferative Response of Chondrocytes to Melatonin in Girls with Adolescent Idiopathic Scoliosis Wei-Jun Wang, Hiu-Yan Yeung, Chi-Wai Man, Kwong-Man Lee, Kin-Wah Ng, Yong Qiu and Jack Chun Yiu Cheng Normal Expression of Melatonin Receptors (MT1, MT2) in BMSCs from Adolescent Idiopathic Scoliosis Patients and Its Significance Haibo Li, Weijun Wang, Guangquan Sun, Jack Chun-Yiu Cheng and Yong Qiu Elevated Plasma Factor P Is Involved in Idiopathic Scoliosis Onset and Curve Progression A. Moreau, A. Franco, B. Azeddine, P.H. Rompré, I. Turgeon, K.M. Bagnall, B. Poitras, H. Labelle, C.-H. Rivard, G. Grimard, J. Ouellet, S. Parent, G. Larouche and G. Lacroix The Effect of Melatonin on Proliferation and Differentiation of Osteoblasts in Adolescent Idiopathic Scoliosis vs Normal Control G.C.W. Man, Y.H. Yeung, W.J. Wang, K.M. Lee, B.K.W. Ng, W.Y. Hung, Y. Qiu and J.C.Y. Cheng A Randomized Controlled Trial on Treatment Outcome and Patient’s Acceptance of the SpineCor vs Rigid Bracing System for AIS Girls T.P. Lam, M.S. Wong, B.K.W. Ng, S.W. Sin, R.H.K. Kwok, W.T.K. Lee, S.L.F. Shum, D.H.K. Chow and J.C.Y. Cheng Is Full Torso Imaging for the Assessment of Torso Deformity in Scoliosis Worth Its Cost? P.O. Ajemba, N.G. Durdle and V.J. Raso Importance of the Immediate Correction for the Effectiveness of Brace Treatment in AIS J. Clin, C.-É. Aubin, S. Parent and H. Labelle

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Subject Index

377

Author Index

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Chapter 1 Genetics

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Chapter 2 Growth

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Body mass index of girls in health influences menarche and skeletal maturation: a leptin-sympathetic nervous system focus on the trunk with hypothalamic asymmetric dysfunction in the pathogenesis of adolescent idiopathic scoliosis? RG BURWELL1, RK AUJLA1, AS KIRBY1, PH DANGERFIELD2, A MOULTON3 AA COLE1, FJ POLAK1, RK PRATT1 J K WEBB1 1

Centre for Spinal Studies and Surgery, Nottingham University Hospitals Trust, Queen’s Medical Centre Campus, Nottingham, UK, 2 The University of Liverpool and Staffordshire University, UK, 3 Department of Orthopaedic Surgery, King’s Mill Hospital, Mansfield, UK (Supported by AO), Abstract. Lower body mass index (BMI) and lower circulating leptin levels have been reported in girls with AIS. In this paper we evaluate skeletal sizes and asymmetries by higher and lower BMI subsets about the means for each of three groups of girls age 1118 years: 1) normals, 2) school screening referrals, and 3) preoperative girls. Higher and lower BMI subsets, likely to have separated subjects with higher from those with lower circulating leptin levels, identify: 1) girls with relatively earlier and later menarche; 2) trunk width size greater in the higher than in the lower BMI subset, of all three groups; 3) abnormal upper arm length (UAL) asymmetries (right minus left) in the lower BMI subset of the preoperative girls; and 4) in thoracic AIS of screened and preoperative girls, Cobb angle and apical vertebral rotation each significantly and positively correlate with UAL asymmetry in the lower BMI subset but not in the higher BMI subset. In preoperative girls, the lower BMI subset shows the combination of relatively reduced pelvic width and abnormal UAL asymmetry, suggesting that both are linked to lower circulating leptin levels. An earlier puberty with hormonal changes provides a plausible explanation for the larger trunk width at the shoulders and pelvis especially at the younger ages in the higher BMI subsets. At the shoulders, this widening is driven by the ribcage which, in human evolution was acquired with decoupling of head and trunk movements required for efficient bipedal gait. The UAL asymmetry patterns within the groups and BMI subsets are not explained by hormonal mechanisms. It is hypothesized that 1) normal trunk widening of the thoracic cage by hormones in human adolescence is supplemented via the sympathetic nervous system under leptin-hypothalamic control influenced by energy stores (metabolic fuel); and 2) hypothalamic dysfunction with altered hypothalamic sensitivity to leptin through a SNS-driven asymmetric effect may create skeletal length asymmetries in upper arms, ribs, ilia and vertebrae, and initiate AIS. Additional mechanisms acting in the spine and

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R.G. Burwell et al. / BMI of Girls in Health Inﬂuences Menarche and Skeletal Maturation trunk may be required for AIS to progress including 1) somatic nervous system dysfunction, 2) biomechanical spinal growth modulation, and 3) osteopenia. Key words.. Idiopathic scoliosis, pathogenesis, spine, body mass index, leptin, shoulder girdle, pelvis, ribcage, human evolution

1. Introduction Lower body mass index (BMI) has been detected in girls with AIS by some [1-7] but not by all [8] and BMI is normal before developing scoliosis [9]. Qiu et al [7] reported that low body mass index in AIS girls is associated with a marked decrease in circulating leptin compared with controls and leptin was found to correlate positively with corrected height. They comment [7] on clinical and experimental evidence strongly suggesting that leptin acts on bone tissue through both central and peripheral pathways; a central pathway driven by hypothalamic nuclei and the sympathetic nervous system (SNS), and peripherally by stimulating osteoblastic differentiation and inhibiting osteoclastic activity, resulting in enhanced bone formation and reduced bone resorption, and that leptin may regulate body growth during childhood and adolescence. There are no other reports on how skeletal size in AIS girls may relate to BMI. In this paper we evaluate skeletal sizes and asymmetries by higher and lower BMI subsets. The findings give some support to our leptin-SNS concept for the pathogenesis of adolescent idiopathic scoliosis [10]. 2. Methods and subjects 2.1 Three groups of subjects Data from three groups of female subjects age 11-18 years were studied: 1) normals (n=274, mean age 13.4 years); 2) school screening referrals (n=137, mean age 14.8 years, mean Cobb angle 15.8 degrees, mean apical vertebral rotation (AVR) 12.6 degrees, thoracic 37, thoracolumbar 41, lumbar 41, double 15, straight 3); and 3) preoperative subjects (n=110, mean age 14.8 years, mean Cobb angle 55.6 degrees, mean apical vertebral rotation 27.1 degrees, thoracic 81, thoracolumbar 25, lumbar 4). The numbers include subjects with a complete set of upper arm lengths, standing height and weight but tibial lengths were not recorded in some of the screened and preoperative girls. The normal subjects were examined by one observer (RGB) in 1973-1981. The scoliosis screening girls were referred to hospital by a routine quantitative protocol [11] and examined by the same observer in 1988-1999. The preoperative subjects were examined by one of three observers (RGB, AAC or RKP) in 1993-1999. 2.2 Anthropometry (Figure 1) The anthropometric techniques utilized the Harpenden Anthropometric Instruments (Holtain Ltd, Crosswell, Crymych, Pembs. SA41 3UF UK)[12,13]. Standing and sitting heights were recorded and subischial height calculated as standing height minus sitting height. Corrected standing and sitting heights for the screened and preoperative girls were calculated with the Bjure-Nachemson Formula [14]. A Harpenden

R.G. Burwell et al. / BMI of Girls in Health Inﬂuences Menarche and Skeletal Maturation

11

anthropometer was used to measure biacromial width, biiliac width, upper arm lengths (UALs) forearm-with-hand lengths and tibial lengths. UAL asymmetry and tibial length asymmetry were calculated as right minus left. An association between AIS thoracic curve AVR and UAL asymmetries was detected in a previous study [15,16]. Tibial length [TL] provided a model for the lower limbs in a previous study [17,18]. Menarcheal age was recorded. 2.3 Anthropometric error studies Intra-observer errors for RGB are (n=126): [19-21]: Standing height: Technical error of measurement (TEM)=2.1mm, coefficient of reliability R=0.99, Sitting height: TEM=1.9 mm R=0.97 Biacromial width: TEM= 3.3 mm Biiliac width: TEM=1.99 mm Upper arm length (right): TEM=2.9 mm, R=0.95, Upper arm length asymmetry: TEM=3.6 mm, R=0.39 Tibial length (right) TEM=3.2 mm, R=0.96 Tibial length asymmetry TEM=3.7mm, R=0.38. 2.4. Body mass index Body mass index (BMI=weight in kg/stature in metres) above and below the BMI mean for each group was used to define two subsets of subjects with higher and lower BMIs. Skeletal parameters were plotted against decimal age for these two BMI subsets of each group, including stature, sitting height, subischial height, biacromial width, biiliac width and for paired structures right and left for each of upper arm length (UAL), forearm-with-hand length, and tibial length (TL). Differences between each skeletal parameter by BMI subset was evaluated by analysis of variance (ANOVA) with age correction. Skeletal length asymmetries of UALs and TLs for the two subsets above and below the mean for each group were evaluated by an independent t-test. (The findings for each skeletal parameter by standard deviation scores will be reported separately). 2.5 Spinal radiographs The spinal radiographs were read by one observer (RGB) for the screened and 42 of the preoperative girls for Cobb angle and apical vertebral rotation (AVR)[22], and for the other preoperative girls by another observer (RKP). The intra-observer error for RGB fpr measuring Cobb angle is TEM=1.3 degrees, R=0.82, n=10; and for AVR, TEM=3.3 degrees, R=0.63, n=10 [15]. 3. Results 3.1 Body mass index. BMI means and standard deviations for the three groups are respectively 19.2±2.7, 19.6±2.7 and 19.6±3.3 and not significantly different between any of the groups

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(analysis of variance, correcting for age). BMI increases significantly with age in each of normals (ρ=0.408, p<0.001). screening referrals (ρ=0.181 p=0.026) and preoperative girls (ρ =0.271, p=0.002)(Spearman rank correlation coefficients). 3.2. General skeletal growth (Figures 1 and 2) and menarche In each of the three groups, normals, screened and preoperative girls, significantly larger size is attained in those subjects in the higher BMI subset than in those in the lower BMI subset with respect to biacromial width and biiliac width (ANOVA with correction for age). Figure 1 for the screened girls shows for the anatomical regions the statistical levels of significance between the higher and lower BMI subsets. In preoperative girls, the biacromial and biiliac widths are significantly larger in the higher BMI subset than in the lower BMI subset (p<0.001), but not in sitting height, upper arm length or tibial length. In the normals, the effect is present in biiliac width (p=0.014) and biacromial width (p=0.0.036) and not in sitting height or upper arm length. The mean age at menarche for the screened girls, is significantly earlier in the higher BMI subset, than in the lower BMI subset (12.5 and 13.1 years, p=0.003, n=51 and 60 respectively) (Figure 2) (overall, premenarcheal 23, postmenarcheal 111, not known in 3). 3.3 Extra-spinal skeletal length asymmetries by higher and lower BMI subsets In asymmetries of upper arm length and tibial length differences by higher and lower BMI subset were calculated by independent t-test and by analysis of variance (ANOVA) without and with correction for age respectively. 3.3.1 Upper arm length asymmetries (Figure 3) a)

Normal girls not significantly different (higher BMI 2.4 mm, lower BMI 2.1 mm, p=0.623, n=131 and 143 respectively), ANOVA p=0.960, p=0.910. b) Screened girls not significantly different (higher BMI 1.3 mm, lower BMI -0.5 mm, p=0.070, n=54 and 83 respectively), ANOVA p=0.062, p=0.146. c) Preoperative girls significantly different (higher BMI 3.4 mm, lower BMI 7.0 mm, p=0.017, n=54 and 56 respectively), ANOVA p=0.063, p=0.503 Figure 3 shows that UAL asymmetries of girls with higher BMI subsets are not significantly different between groups but for lower BMI subsets are significantly different between the preoperative and each of the screened and normal girls (each p<0.001). 3.3.2 Tibial length asymmetries a) Normal girls not significantly different (higher BMI -0.2 mm, lower BMI -0.3 mm, p=0.805, n=131 and 142 respectively), ANOVA p=0.486, p=0.570. b) Screened girls not significantly different (higher BMI 1.4 mm, lower BMI 2.4 mm p=0.214, n=55 and 78 respectively)., ANOVA p=0.227, p=0.770. c) Preoperative girls not significantly different (higher BMI 0.4 mm, lower BMI 1.1 mm, p=0.561, n=41 and 44 respectively), ANOVA p=0.973, p=0.905.

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Figure 1. Photographs of six anthropometric techniques and a drawing for scoliosis screened girls showing statistical findings by analysis of variance with correction for age. Some skeletal regions show relatively larger sizes for the higher than for the lower BMI subset with p-values given; these are: biacromial width, biiliac width, upper arm length and sitting height. Findings are shown only for right limbs. A similar pattern of trunk features is present in normal and preoperative girls at different levels of statistical significance.

Figure 2. Scoliosis screened girls. Graph showing best-fit quadratic regression lines for biiliac widths by age in years for girls in the higher and lower BMI subsets. The girls in the higher BMI subset have relatively larger biiliac widths than the girls in the lower BMI subset especially at the younger ages. (p=0.001, analysis of variance with correction for age). Mean menarcheal ages are shown for the higher BMI (MH) and lower BMI (ML) subsets.

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Figure 3. Plots of mean asymmetries for upper arm length (UAL mm. right minus left) for the three groups – normal, screened and preoperative girls by higher and lower BMI subsets; the numbers beneath the means show the number by group in each subset and below the mean Cobb angle (CA). BMI=body mass index, H=higher BMI subset, L=lower BMI subset. The statistical significances levels for the comparisons are indicated above the horizontal bars (ANOVA with correction for age). Note, that the mean UAL asymmetries associated with the higher BMI subset (upper block) are not significantly different for any of the comparisons, while the UAL asymmetry associated with the preoperative lower BMI subset (lower block) is significantly more than that of the screened and normal girls [see 10].

3.4. Thoracic curves: Cobb angles and AVRs 3.4.1 Mean values of AVR and Cobb angles In thoracic AIS of screened and preoperative girls (to provide a range of curve severities) absolute AVR (but not absolute Cobb angle) is significantly larger in the higher BMI subset than in the lower BMI subset (mean AVR 23.3 degrees and 19.8 degrees, p=0.026 independent t-test; mean Cobb angle 45.4 degrees and 43.9 degrees,

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p=0.680 independent t--test, n=57, with 6 left curves, and n=61 with 6 left curves respectively). Comparing the higher with the lower BMI subsets by ANOVA with age correction for AVR p= 0.811 and for Cobb angle p=0.944. 3.4.2. AVR and Cobb angle are each associated with upper arm length asymmetry. In the same group of thoracic AIS screened and preoperative girls, in the lower BMI subset, but not in the higher BMI subsets, both AVR and Cobb angle correlate significantly with UAL length asymmetry – lower BMI subset AVR r=0.459 p<0.001; Cobb angle r=0.526, p<0.001 n=61; higher BMI subset AVR r=0.145, p=0.287; Cobb angle r=0.032, p=0.857, n=57.. Comparing the higher with the lower BMI subsets by ANOVA without age correction for AVR p=0.001 and for Cobb angle p=0.102, and with age correction for AVR p= 0.689 and for Cobb angle p=0.655. 4. Discussion 4.1. Body mass index. The mean BMIs for screened and preoperative girls (each 1990s) are not different from the mean BMI for normals (1973-1981). Using normal BMI findings obtained in the mid-1990s (Aujla RK and Kirby AS, unpublished observations) the screened, but not the preoperative, girls have significantly lower BMIs (p=0.014 and p=0.230 respectively). 4.2. General skeletal growth features, focus on the trunk and relatively earlier skeletal maturation especially of trunk width in subjects with higher BMIs 4.2.1. Broadening of the human shoulder girdle held on the ribcage (Figure 1). The higher and lower BMI subsets are likely to have separated subjects with higher from those with lower circulating leptin levels [7]. Supposed higher circulating leptin levels in all three groups are associated with relatively larger growth in the trunk biacromial and biiliac widths and statistically most evident in the preoperative girls. Contrariwise, supposedly lower circulating leptin levels are associated with relatively less growth in trunk widths. Biacromial width reflects the shoulder girdle which is held on the broad human thoracic cage [23]; the width of the shoulder girdle in human evolution was acquired with the decoupling of head and trunk movements [24,25] required for efficient bipedal gait [26]. Hence, biacromial widths are evidently associated with the lateral growth of the human thoracic cage and clavicles; and our findings suggest that the growth of both widths may be affected by circulating leptin levels in humans but may not in non-human primates and quadrupeds. 4.2.2. Relatively earlier menarche and relatively earlier skeletal maturation in the biacromial width and pelvic width with higher BMIs In the screened girls, the relatively earlier menarche by about 6 months of the higherto- lower BMI subset respectively is in keeping with knowledge of a link between

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body fat and the timing of puberty with leptin playing a permissive role [27,28] and kisspeptin activation of the G-protein coupled receptor-54 [28-31] providing a critical metabolic signal initiating puberty through pulsatile GnRH secretion [29]. Hence, relatively earlier skeletal maturation in the higher BMI subset due to relatively earlier puberty with hormonal changes provides a plausible explanation for the trunk width differences by BMI subset at the shoulder (pectoral) girdle) and pelvis. The adult human pelvis is wider than it is high [32]. 4.2.3. How to explain the skeletal asymmetries – trunk width and human bipedal gait While this hormonal interpretation is a plausible explanation for the relative differences in trunk width growth by higher and lower BMI subsets, it does not explain: a) The upper arm length (UAL) asymmetry patterns which differ between preoperative and each screened and normal girls (Figure 3). This UAL asymmetry effect is evident in the preoperative lower BMI subset composed of taller and thinner girls, in whom trunk width growth is reduced relative to higher BMI girls suggesting both are linked to lower circulating leptin levels. b) In thoracic AIS, Cobb angle and apical vertebral rotation are each associated with upper arm length (UAL) symmetry in the lower, but not the higher, BMI sunset. These patterns with respect to UAL asymmetry can be explained by postulating asymmetry from asynchrony in the upper arm growth plates from intrinsic or extrinsic mechanisms. There is no evidence for an intrinsic mechanism, but there is clinical and experimental evidence to suggest that a SNS-driven asymmetry mechanism in right thoracic AIS in girls determines concave periapical rib relative overgrowth [33-35] and in AIS of the lower spine (thoracolumbar and lumbar curves) determines the concave ilium relative overgrowth [36]. If this putative SNS-driven effect may create skeletal asymmetries, it may also supplement the hormonal contribution to trunk width by an SNS-driven general effect on trunk width growth. This SNS effect may be particular to humans and, as already stated, at the shoulders be driven by the ribs in a process that evolved with decoupling of head and trunk movements [24,25] required for efficient bipedal human gait [26,37]. 4.3. A leptin-sympathetic nervous system (SNS) effect on biacromial widths?

and pelvic

We propose the hypothesis that the hormonally-driven skeletal width growth in the human trunk is supplemented by a normal leptin-hypothalamic-SNS-driven mechanism related to energy stores (metabolic fuel) with leptin as a signal to the hypothalamus. In general, the coupling of skeletal growth to energy balance [38,39] involves leptin and Y2-receptors on neuropeptide Y (NPY-ergic) hypothalamic neurons [40,41 ]. . 4.4. Hypothalamic dysfunction and altered sensitivity to circulating leptin? In the lower BMI subset of the screened with preoperative girls with thoracic AIS, larger Cobb angles and larger AVRs are each associated with a longer right-to-left

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upper arms. This pattern suggests a mechanism linking each of 1) circulating leptin levels, 2) growth of humeri, ribs and vertebrae, and 3) development of the scoliosis deformity. In the preoperative girls, the presence of abnormal UAL asymmetry with the lower BMI subset (Figure 3) is consistent with a putative dysfunction of the hypothalamus [10] with stress in response to lower circulating leptin levels and expressed as ‘upper arm wide asymmetry’, defined as affecting upper arms, ribs and vertebrae. The absence in preoperative girls of abnormal UAL asymmetry with the higher BMI subset suggests a protective effect of circulating leptin levels on the dysfunctional hypothalamus, with the breadth of the induced skeletal asymmetry being limited to the ribs and/or spine as ‘trunk wide asymmetry’, defined as affecting spine and/or ribs but not humeri. For the spine these concepts may be evaluated, in part, by combined MRI and histological studies of periapical vertebral growth plates [42] in relation to BMI. 4.5. Tibial length asymmetries The absence of tibial length asymmetries being significantly associated with higher and lower BMI subsets suggests a focus on the trunk of girls for the putative SNSdriven effect that may determine the asymmetries of each of UAL, periapical ribs and spine. 4.6. SNS-driven asymmetries in 3D? The postulated SNS-driven effects on growing bones may exert their asymmetries in the spine in 3D. Sagittal plane severity of thoracic AIS needs to be evaluated by higher and lower BMI subsets. 5. Other mechanisms in curve progression In addition to the hormonal and putative leptin-hypothalamic-SNS-driven effects on certain growing bones (humeri, ribs and vertebrae) in the screened and preopertative girls, we suggest that somatic nervous system dysfunction (? Relative postural maturational delay) may also be needed for progression of AIS curves to the severity requiring surgery [37,43]. 6. Conclusions 1. In terms of general skeletal growth, significantly larger size in trunk widths (biacromial and biiliac) is attained in girls in the higher BMI subset than in those in the lower BMI subset.. 2. Higher and lower BMI subsets are likely to have separated girls with elevated from those with reduced circulating leptin levels. 3. In the screened girls, the relatively earlier menarche by about 6 months of the higher–to-lower BMI subset is in keeping with knowledge of a link between body fat and the timing of puberty with leptin as a signal playing a permissive role [27,28]. 4. Relatively earlier skeletal maturation in the higher BMI subset due to an earlier puberty with hormonal changes provide a plausible explanation for the relatively larger

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trunk width at the shoulders and pelvis. 5. This interpretation does not explain the abnormal upper arm length (UAL) asymmetry differences in the lower BMI subset between preoperative and each of screened and normal girls (Figure 3). Nor does it explain in thoracic AIS in the lower BMI subset the statistically significant association between each of Cobb angle and AVR with UAL asymmetry and not in the higher BMI subset. 6. In the lower BMI subset composed of taller and thinner girls, UAL asymmetry is most evident in the preoperative group (Figure 3), and in whom trunk width growth for age is reduced relative to the higher BMI girls, suggesting that both UAL asymmetry and the relatively reduced trunk width growth are linked to lower circulating leptin levels. 7. These UAL asymmetries can be explained by postulating asymmetry from asynchrony in the upper arm growth plates from intrinsic or extrinsic mechanisms. There is no evidence of mechanisms intrinsic to humeral growth plates but clinical and experimental evidence [10] suggests that a SNS-driven asymmetric mechanism in AIS girls may determine concave periapical rib relative overgrowth in right thoracic AIS in girls [33-35] and concave ilium relative overgrowth in lower spine AIS (thoracolumbar and lumbar curves) [36]. 8. If this putative SNS-driven asymmetric effect can create such skeletal length asymmetries, it may also supplement the hormonal contribution to trunk width growth 9. We propose the hypothesis that the hormonal stimulus to normal human trunk width growth is supplemented by a normal leptin-hypothalamic-SNS-driven stimulus related to energy stores (metabolic fuel) with leptin as a signal to the hypothalamus [10]. 10. Biacromial width reflects the breadth of the human thoracic cage [23]. The width of the shoulder (pectoral) girdle was acquired in human evolution with decoupling of the head and trunk movements [24,25] required for efficient bipedal gait [26], as was pelvic widening [32]. 11. It is suggested that the thoracic AIS of screened and preoperative girls was initiated by dysfunction of a normal hypothalamic control of trunk width growth associated with altered hypothalamic sensitivity to leptin (Figure 3). 12. Our hypothesis for AIS in girls states that [10] given adequate nutrition and energy stores, circulating leptin talks to the hypothalamus where dysfunction leads to an altered sensitivity to leptin resulting in increased SNS activity contributing with neuroendocrine mechanisms to: 1) earlier age at, and increased peak height velocity, 2) general skeletal overgrowth, 3) earlier skeletal maturation, 4) extra-spinal skeletal length asymmetries, including periapical ribs, and the ilia, 5) generalized osteopenia, and 6) lower BMI. 13. In AIS girls, a dysfunctional hypothalamus under stress from lower circulating leptin levels, expresses skeletal length asymmetry through the SNS in the upper arms, ribs and spine (‘upper arm wide asymmetry’) but, with high circulating leptin levels, is not expressed in the upper arms (‘trunk wide asymmetry’) where the asymmetry is limited to vertebrae and/or ribs. 14. In addition to the hormonal and the putative leptin-hypothalamic-SNS-driven asymmetric effects [10] that may initiate AIS in girls, other mechanisms required for curve progression may include 1) somatic nervous system dysfunction (? Relative postural maturational delay) [37,43], 2) spinal growth modulation [44], and 3) osteopenia. [45].

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15. Evidence suggests that right thoracic AIS in females is initiated by the sympathetic nervous system [33-35]. Left thoracic AIS in females in a pilot study showed MRI changes in the somatic nervous system (cerebral hemispheres and corpus callosum)[46]. Does this imply different relative contributions of the autonomic and somatic nervous systems to the pathogenesis of right and left thoracic AIS respectively in females? 16. Compared with the brains of non-scoliotic girls, may the brains of girls who develop AIS have other differences of asymmetric functions [47] some sexually dimorphic and possibly linked to raised testosterone levels [10], of which the scoliosis is a solitary expression? References [1] [2]

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[17] Burwell RG, Aujla RK, Freeman BJC, Dangerfield PH, Cole AA, Kirby AS, Pratt RK, Webb JK, Moulton A: Patterns of extra-spinal left-right skeletal asymmetries and proximo-distal disproportion in adolescent girls with lower spine scoliosis: ilio-femoral length asymmetry and bilateral tibial-foot length disproportion. In: Research into Spinal Deformities 5, Studies in Health Technology and Informatics Volume 123. Edited by: Uyttendaele D, and Dangerfield PH, Amsterdam, IOS Press; 2006: 101-108. [18] Burwell RG, Aujla RK, Freeman BJC et al, Proximo-distal skeletal length disproportion in lower limbs of girls with adolescent idiopathic scoliosis (AIS) compared with normal girls: tibial length/foot length is larger bilaterally and associated with left-right tibial length asymmetry and scoliosis Clin Anat 2007, 20(4):467. [19] Burwell RG, Vernon CL, Dangerfield PH. Chapter 38, Skeletal measurement, In Scientific Foundations of Orthopaedics and Traumatology, Edited by Owen R, Goodfellow J, Bullough P, London: William Heinemann Medical Books Limited, 1980, 317-329. [20] Cole AA: Quantitation of scoliosis before and after surgery. DM thesis, University of Nottingham, UK, 2005. [21] Ulijasek SJ, Mascie-Taylor CGN, Chapter 3, Intra- and inter-observer error in anthropometric measurement. In Anthropometry: the individual and the population. Cambridge:University Press, 1994, 30-55. [22] Perdriolle R, La Scoliose, son etude tridimensionelle. Maloine SA Editeur, 27 Rue de l’Ecole-deMedicine, 75005, Paris 1979. [23] Aiello L, Dean C, Chapter 17, The hominoid arm. In, An Introduction to Human Evolutionary Anatomy, London: Academic Press 1990, pp 342-371. [24] Alemseged Z, Spoor F, Kimbel WH, Bobe R, Geraads D, Reed D, Wynn JG: A juvenile hominid skeleton from Dikika, Ethiopia. Nature 2006, 443:296-301. [25] Wong K. Lucy’s baby. An extraordinary new human fossil renews debate over the evolution of upright walking. Sci Amer 2006, 295(6):56-63. [26] Bramble DM, Lieberman DE. Endurance running and the evolution of Homo. Nature 2004, 432:345352. [27] Ebling FJP, The neuroendocrine timing of puberty. Reproduction 2005, 129:675-683. [28] Kaplowitz PB, Link between body fat and the timing of puberty. Pediatrics 2008, 121, Supplement S208-S217. [29] Crown A, Clifton DK, Steiner RA. Neuropeptide signaling in the integration of metabolism and reproduction. Neuroendocrinol 2007, 86(3):175-182. [30] Kauffman AS, Clifton DK, Steiner RA, Emerging ideas about kisspeptin-GPR54 signaling in the neuroendocrine regulation of reproduction. Trends Neurosci 2007, 30(10):504-51. [31] Plant TM, The role of KISS-1 in the regulation of puberty in higher primates. Eur J Endocrinol 2006, 155 Suppl 1:511-516. [32] Aiello L, Dean C, Chapter 20, The Hominoid pelvis, In, An Introduction to Human Evolutionary Anatomy, London: Academic Press 1990, pp429-456. [33] Sevastik JA: The thoracospinal concept of the pathogenesis of idiopathic scoliosis. In Etiology of Adolescent Idiopathic Scoliosis: Current Trends and Relevance to New Treatment Approaches, Edited by Burwell RG,.Dangerfield PH, Lowe TG, Margulies JY. State of the Art Reviews: Spine 2000, 14(2):391-400, Philadelphia, Hanley & Belfus Inc. [34] Sevastik J, Burwell RG, Dangerfield PH: A new concept for the etiopathogenesis of the thoracospinal deformity of idiopathic scoliosis: summary of an electronic focus group debate of the IBSE. Eur Spine J 2003. 12:440-450. [35] Sevastik J.A: Right convex thoracic female adolescent scoliosis in the light of the thoracospinal concept. In Research into Spinal Deformities 5, Studies in Health Technology and Informatics Volume 123. Edited by Uyttendaele D, Dangerfield PH, Amsterdam, IOS Press; 2006:552-558. [36] Burwell RG, Aujla RK, Freeman BJC et al, Patterns of extra-spinal left-right skeletal asymmetries in adolescent girls with lower spine scoliosis: relative lengthening of the ilium on the curve concavity and of right lower limb segments. In Research into Spinal Deformities 5, Studies in Health Technology and Informatics Volume 123. Edited by Uyttendaele D, Dangerfield PH, Amsterdam, IOS Press; 2006: 5765. [37] Burwell RG, Dangerfield PH, Freeman BJC: Concepts on the pathogenesis of adolescent idiopathic scoliosis. Bone growth and mass, vertebral column, spinal cord, brain, skull, extra-spinal left-right skeletal length asymmetries, disproportions and molecular pathogenesis. In 1st SOSORT Instructional Course Lecture Book. The conservative scoliosis treatment, Studies in Health Technology and Informatics Volume 135, Edited by Grivas TB, Amsterdam IOS Press, 2008, 3-52.

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[38] Gat-Yablonski, G, Phillip M, Leptin and regulation of linear growth. Curr Opin Clin Nutr Metab Care 2008, 11(3):303-308. [39] Iwaniec UT, Boghossian S, Lapke PD et al, Central leptin gene therapy corrects skeletal abnormalities in leptin-deficient ob/ob mice Peptides 2007, 28:1012-1019. [40] Allison SJ, Baldock PA, Herzog H, The control of bone remodeling by neuropeptide Y receptors Peptides 2007, 28: 320-325. [41] Baldock PA, Allison SJ, Lundberg P et al, Novel role of Y1 receptors in the coordinated regulation of bone and energy. J Biol Chem 2007, 282(26):19092-19102. [42] Day G, Frawley K, Phillips B et al, The vertebral body growth plate in scoliosis: a primary disturbance in growth? Scoliosis 2008;3:3 doi 10.1186/1748-7161-3-3. [43] Burwell RG, Dangerfield, PH, Freeman BJC et al,, Etiologic theories of idiopathic scoliosis. Somatic nervous system and the NOTOM escalator concept as one component of the pathogenesis of adolescent idiopathic. This Meeting. [44] Stokes IAF: Analysis and simulation of progressive adolescent scoliosis by biomechanical growth modulation. Eur Spine J 2007, 16:1621-1628. [45] Lee WT, Cheung CS, Tse YK et al, Association of osteopenia with curve severity in adolescent idiopathic scoliosis: a study of 919 girls. Osteopor Intern 2005, 16(12):1924-1932 [46] Chu W, Shi L, Wang D, Paus T et al, Anatomical difference of brains in subjects with right thoracic and left thoracic adolescent idiopathic scoliosis (AIS): MR-based morphometric study [abstract]. In Proceedings of Britspine, Fifth Combined Meeting, 30th April-2nd May 2008, Belfast Waterfront Hall, Belfast, Northern Ireland. [47] Goldberg CJ, Dowling FE, Fogarty EE et al, Adolescent idiopathic scoliosis and cerebral asymmetry. Examination of a nonspinal perceptual system. Spine 1995, 20(15):1685-1691.

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Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved. doi:10.3233/978-1-58603-888-5-22

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Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved. doi:10.3233/978-1-58603-888-5-29

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Back trunk morphology in 3301 children aged 3-9 years old T B. GRIVAS, E S. VASILIADIS, C MIHAS, C MAZIOTOU, G TRIANDAFYLLOPOULOS Scoliosis Clinic, Orthopaedic Department, “Thriasio” General Hospital, G. Genimata Avenue, Magula, 19600, Athens, Greece. Abstract. The present cross sectional study reveals trunk asymmetry (TA) in "normal" Mediterranean juveniles for the first time. The scoliometer readings in both standing and sitting forward bending position (FBP) of 3301 children, (1645 boys, and 1656 girls) aged from 3 to 9 years old were studied. TA was quantified by measuring angle of trunk rotation (ATR) and children were divided in two groups. In group I the ATR was 1° to 6° degrees and in group II ≥7°. 71.25% of boys and 73.27% of girls in standing while 81.13% of boys and 80.74% of girls in sitting FBP, were symmetric (ATR = 0o). The symmetry difference at standing minus sitting FBP for boys and girls was 9.88% and 7.43% respectively. Severe asymmetry (ATR≥7°) was found in 1.74% of boys and in 1.75% of girls at the standing and in 1.21% and 1.22% at the sitting FBP respectively. Analysing ATR by age it appears that significant TA changes occur between 8 - 9 years of age for boys and between 6-7 and 8 - 9 years for girls. The amount of trunk asymmetry in children is the indicator for referral and further orthopaedic assessment. This report provides, for the first time information about the variability of back morphology in “normal” juveniles which is worth knowing when a child is examined for juvenile scoliosis. Keywords. Juvenile idiopathic scoliosis, juveniles, back morphology, trunk asymmetry.

1.

Introduction

Trunk asymmetry (TA) is considered by many authors as the clinical presentation of scoliosis [1, 2, 3]. Although the incidence of scoliosis in juveniles is much smaller than adolescents, in younger girls referred from a school-screening program there was a discrepancy between the thoracic asymmetry and the morphology of the spine [4]. Twenty five per cent of children with Angle of Trunk Rotation (ATR) ≥ 7o had either a straight spine or a curve smaller than 10o [4], which means that in juveniles, trunk asymmetry is not a sensitive clinical sign for scoliosis. As a baseline for further research on this observation, we need to know what the incidence of trunk asymmetry in a normal juvenile population is. In a previous study we reported trunk asymmetry in normal adolescents [5]. The present cross sectional study quantifies for the first time trunk asymmetry in "normal" Mediterranean juveniles. 2.

Method and Material

The scoliometer readings in both standing and sitting forward bending position (FBP) of 3301 children, (1645 boys, and 1656 girls) aged from 3 to 9 years old were studied. TA was quantified

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T.B. Grivas et al. / Back Trunk Morphology in 3301 Children Aged 3–9 Years Old

by measuring angle of trunk rotation (ATR) in the thoracic, thoracolumbar and lumbar regions of the back. Children were divided in three groups. In group 0 children were symmetric, in group 1 the ATR was 1°-6° (moderate asymmetry) and in group 2 the ATR was ≥7° (severe asymmetry). Frequency of symmetry, moderate and severe asymmetry for both boys and girls was quantified in standing and in sitting FBP for all the examined regions of the back. The difference of trunk asymmetry between standing and sitting FBP as well as differences between boys and girls in frequency of trunk asymmetry were also calculated. 3.

Results

74.2 % of boys and 77% of girls were symmetric (ATR=0o) in the thoracic region in standing FBP, while 82.7% of boys and 84.1% of girls were symmetric in the thoracic region in sitting FBP. Symmetry and asymmetry in all the three examined regions of the spine in the standing FBP is shown for boys in Table 1 and for girls in Table 2, while in the sitting FBP it is shown for boys in Table 3 and for girls in Table 4.

Thoracic

Thoraco lumbar

Lumbar

Symmetry

1

-1

2

-2

Missing

Total

N

1209

264

142

11

3

16

1645

%

74.22

16.21

8.72

0.68

0.18

N

1155

275

168

22

9

%

70.90

16.88

10.31

1.35

0.55

N

1118

302

169

28

12

%

68.63

18.54

10.37

1.72

0.74

100.00 16

1645 100.00

16

1645 100.00

Table 1: Frequency of symmetry and asymmetry in boys. Scoliometer readings are obtained at standing forward bending position. 1: mild right asymmetry (ATR 1-6o), 2: severe right asymmetry (ATR ≥7o), -1 mild left asymmetry (ATR 1-6o), -2: severe left asymmetry (ATR ≥7o).

Thoracic

Thoraco lumbar

Lumbar

Symmetry

1

-1

2

-2

Missing

Total

N

1262

221

139

8

9

17

1656

%

77.00

13.48

8.48

0.49

0.55

N

1185

254

168

19

13

%

72.30

15.50

10.25

1.16

0.79

N

1156

255

191

25

12

%

70.53

15.56

11.65

1.53

0.73

100.00 17

1656 100.00

17

1656 100.00

Table 2: Frequency of symmetry and asymmetry in girls. Scoliometer readings are obtained at standing forward bending position. 1: mild right asymmetry (ATR 1-6o), 2: severe right asymmetry (ATR ≥7o), -1 mild left asymmetry (ATR 1-6o), -2: severe left asymmetry (ATR ≥7o).

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T.B. Grivas et al. / Back Trunk Morphology in 3301 Children Aged 3–9 Years Old

Thoracic

Thoraco lumbar

Lumbar

Symmetry

1

-1

2

-2

Missing

Total

N

1348

143

128

6

4

16

1645

%

82.75

8.78

7.86

0.37

0.25

N

1335

144

129

13

8

%

81.95

8.84

7.92

0.80

0.49

N

1282

182

137

19

9

%

78.70

11.17

8.41

1.17

0.55

100.00 16

1645 100.00

16

1645 100

Table 3: Frequency of symmetry and asymmetry in boys. Scoliometer readings are obtained at sitting forward bending position. 1: mild right asymmetry (ATR 1-6o), 2: severe right asymmetry (ATR ≥7o), -1 mild left asymmetry (ATR 1-6o), -2: severe left asymmetry (ATR ≥7o).

Thoracic

Thoraco lumbar

Lumbar

Symmetry

1

-1

2

-2

Missing

Total

N

1378

136

116

6

3

17

1656

%

84.08

8.30

7.08

0.37

0.18

N

1315

159

141

12

12

%

80.23

9.70

8.60

0.73

0.73

N

1277

179

156

17

10

%

77.91

10.92

9.52

1.04

0.61

100.00 17

1656 100.00

17

1656 100.00

Table 4: Frequency of symmetry and asymmetry in girls. Scoliometer readings are obtained at sitting forward bending position. 1: mild right asymmetry (ATR 1-6o), 2: severe right asymmetry (ATR ≥7o), -1 mild left asymmetry (ATR 1-6o), -2: severe left asymmetry (ATR ≥7o).

The difference in the frequency of asymmetry between standing and sitting FBP, in all the examined regions of the spine, for boys and girls are shown in table 5.

Boys Symmetry ATR=0o

ATR 1o- ATR ≥ 7o 6o

Symmetry ATR=0o

Girls ATR 1o6o

ATR ≥ 7o

Thoracic

Thoracolumbar Lumbar

8,5%

8,3%

0,4%

7,1%

6,6%

0,5%

11%

10,4%

0,6%

7,9%

7,5%

0,5%

10,1%

9,3%

0,7%

7,4%

6,8%

0,6%

Table 5. The difference (%) of the frequency of asymmetry between standing and sitting FBP, in the three examined regions of the spine, for both boys and girls.

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4.

T.B. Grivas et al. / Back Trunk Morphology in 3301 Children Aged 3–9 Years Old

Discussion

It is axiomatic, that any screening procedure for abnormality is based on knowledge of normality. Normal juvenile girls appear to be more symmetric than juvenile boys, a finding which is opposite in adolescents [5, 6]. Juveniles appear to have a smaller frequency of trunk asymmetry than adolescents. Furthermore, in the present study the frequency of asymmetry was detected in greater percentage in the standing than in the sitting FBP, the same as in adolescents, which implies leg length or pelvic difference in pathogenesis of trunk asymmetry. The amount of trunk asymmetry in children is the indicator for referral and further orthopaedic assessment if a spinal curve is detected. This report provides, for the first time information about the variability of back morphology in “normal” juveniles which is worth knowing when a child is examined for juvenile scoliosis. References [1] [2] [3] [4]

[5] [6]

Nissinen MJ, Heliövaara MM, Seitsamo JT, Könönen MH, Hurmerinta KA, Poussa MS, Development of trunk asymmetry in a cohort of children ages 11 to 22 years. Spine 25(5) (2000), 570–74. Vercauteren M, van Beneden M, Verplaetse R, Croebe P, Uyttendale D, Verdonk R, Trunk asymmetries in a Belgian school population. Spine 7 (1982), 555–62. Burwell RG, James NJ, Johnson F, Webb JK, Wilson, Standardised trunk asymmetry scores. J Bone Joint Surg 65B (1983), 452–63. YG Grivas TB, Vasiliadis ES, Mihas C, Savvidou OD, The effect of growth on the correlation between the spinal and rib cage deformity. Implications on idiopathic scoliosis pathogenesis. Scoliosis 2(1) (2007), 11. Grivas TB, Vasiliadis E, Koufopoulos G, Segos D, Triantafyllopoulos G, Mouzakis, Study of trunk asymmetry in normal children and adolescents. Scoliosis 1(1) (2006), 19. V Grivas TB, Vasiliadis, Polyzois VD, Mouzakis V, Trunk asymmetry and handedness in 8245 school children. Pediatric ES Rehabilitation, 9(3) (2006), 259-266.

Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved. doi:10.3233/978-1-58603-888-5-33

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The role of the intervertebral disc in correction of scoliotic curves. A theoretical model of Idiopathic Scoliosis pathogenesis T B. GRIVAS, E S. VASILIADIS, G RODOPOULOS, N BARDAKOS Scoliosis Clinic, Orthopaedic Department, “Thriasio” General Hospital, G. Genimata Avenue, Magula, 19600, Athens, Greece. e-mail: [email protected] Abstract. Wedging of the scoliotic inter-vertebral disc (IVD) was previously reported as a contributory factor for progression of idiopathic scoliotic (IS) curves. The present study introduces a theoretical model of IVD’s role in IS pathogenesis and examines if, by reversing IVD wedging with conservative treatment (full- and night-time braces and exercises) or fusionless IS surgery with staples, we can correct the deformity of the immature spine. The proposed model implies the role of the diurnal variation and the asymmetric water distribution in the scoliotic IVD and the subsequent alteration of the mechanical environment of the adjacent vertebral growth plates. Modulation of the IVD by applying corrective forces on the scoliotic curve restores a close-to-normal force application on the vertebral growth plates through the Hueter-Volkmann principle and consequently prevents curve progression. The forces are now transmitted evenly to the growth plate and increase the rate of proliferation of chondrocytes at the corrected pressure side, the concave. Application of appropriately directed forces, ideally opposite to the apex of the deformity, likely leads to optimal correction. The wedging of the elastic IVD in the immature scoliotic spine could be reversed by application of corrective forces on it. Reversal of IVD wedging is thus amended into a “corrective”, rather than “progressive”, factor of the deformity. Through the proposed model, treatment of progressive IS with braces, exercises and fusionless surgery by anterior stapling could be effective. Keywords. Idiopathic scoliosis, intervertebral disc, asymmetrical growth, pathogenesis of idiopathic scoliosis, conservative treatment of Idiopathic scoliosis, fusionless surgery of Idiopathic scoliosis, stapling of the spine.

1.

Introduction

The rationale for management of idiopathic scoliosis (IS) during skeletal growth assumes a biomechanical mode of deformity progression, based on the HueterVolkmann principle [1], whereby extra axial compression decelerates growth and reduced axial compression accelerates it [2]. In treating IS conservatively, application of corrective forces does nothing more than exploiting this principle, by applying appropriately directed forces through the skin, soft tissues and ribs to the vertebral growth plates. The role of the IVD as a contributory factor to the development of the scoliotic curve has been emphasized in a previous study [3]. The response of IVD to abnormal stresses imposed on them in scoliosis is essential to the long-term prognosis of untreated lumbar and thoracolumbar curves [4] and it is very likely that the changes in cartilagenous endplate (vertebral body growth plate) and IVD are key factors in the progression of scoliosis and the manner in which the curve will respond to different therapeutic regimens [5].

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T.B. Grivas et al. / The Role of the Intervertebral Disc in Correction of Scoliotic Curves

The present study illustrates the effect of IVD modulation and its subsequent benefits in IS treatment. The proposed model is examined on conservative treatment (full- and night-time braces and exercises) and fusionless IS surgery with staples. 2.

Material and Methods

A theoretical model of IVD’s role in progressive IS pathogenesis is introduced. The IVD contains the aggrecans of glycosaminoglycans (GAGs) which imbibe water through the so called Gibbs-Donnan mechanism. The highest concentration of GAGs is in the nucleus pulposus (NP) where they are entrapped in a type II collagen network [6] and are exposing a convex-wise asymmetrical distribution. There is an increased collagen content in the NP of the apical IVD but also in the adjacent discs in IS, which is maximal at the apex of the curvature. Furthermore, in the scoliotic spine the NP in the IVD is displaced towards the convex side of the wedged interspaces [4]. Differences also exist in the collagen distribution between the concave and convex sides of the scoliotic annulus fibrosus in IS, with fewer collagen fibres in the concave compared to the convex side [7]. A diurnal variation in the water content of lumbar IVD has also been documented [8, 9], resulting in diurnal variations in loading of the vertebral growth plates, because of IVD’s periodical “swelling” (during night time) and “shrinkage” (for the period of day time under the application of the body load during the upright posture) during the 24 hour period. This asymmetrical pattern of water distribution in the scoliotic IVD, combined with the diurnal variation in the water content of IVDs resulting to a sequence of “swellings” and “shrinkages”, imposes asymmetrical, convex-wise, concentrated cyclical loads to the IVD and the adjacent immature vertebrae growth plates of the child during the 24 hours period [3]. The convex side of the wedged IVD sustains greater amount of cyclic (swelling) expansion than the concave side, leading to the sequelae of asymmetrical growth of adjacent vertebrae (Hueter-Volkmann’s law). Consequently, the response of bone growth to asymmetrical loading follows an asymmetrical pattern and gradually can enhance the correction of the deformity if optimal corrective forces are applied, with full time and night time braces, exercises and fusionless surgery. 3. Results The IVD may be modulated by applying corrective forces on the curve thereby eliminating any asymmetrical accumulation of water in the apical and adjacent IVDs. This, in turn, restores a close-to-normal force application on the vertebral growth plates through the Hueter-Volkmann principle and consequently may prevent curve progression. The forces are now transmitted evenly to the growth plate increasing the rate of proliferation of chondrocytes at the corrected pressure side, the concave. All the stated treatment methods aim at alteration of the mechanical environment and modulation of the endochondral growth of the immature vertebrae. Application of appropriately directed forces, ideally opposite to the apex of the deformity, likely leads to optimal correction. Exercises for IS are using techniques to correct and maintain the correction which has been achieved, during every day activities. By activating the appropriate muscle groups, corrective forces are transmitted on the wedged IVD and are reversing to a degree the wedging, altering the mechanical environment of the adjacent vertebral growth plates.

T.B. Grivas et al. / The Role of the Intervertebral Disc in Correction of Scoliotic Curves

35

Full time braces, through the 3D correction and the combination of derotation together with the three point pressure principle, are exerting constant corrective forces on the wedged IVD and are restoring a close to normal force application on the vertebral growth plates. The mechanical stimulus to the concave side of the growth plate is weaker, allowing new cells to be produced in the proliferateve zone and extracellular matrix to be produced in the hypertrophic zone of the growth plate, according to Hueter – Volkmann principle. As a consequence, longitudinal growth is faster at the concave side of the curve, when a skeletally immature scoliotic child is treated with a full time brace and eventually this could stop its progression or even correct the curve, depending on child’s growth potential. The night time braces are rather overcorrecting the mild or moderate scoliotic curves, simulating gravity, acting also on the apical and adjacent wedged IVDs [10]. Their action is by reducing the previously described asymmetrically imbibed water (greater amount in the convex rather that in the concave side), but additionally they are taking advantage of the diurnal variation in the water content of IVD, which is increased during the night. Hypercorrection of the IVD with the nighttime brace results in vertebral growth under more normal conditions. Under the action of the nighttime brace, the convex side sustains no greater amount of expansion than the concave side, (ceasing the asymmetrical application of Hueter - Volkmann law), reversing the deleterious hypothesis of progression of IS curves; consequently, the growth of the apical and adjacent immature vertebrae turns more normal, within a close - to - normal biomechanical environment. Fusionless surgery with staples, theoretically allows preservation of growth, motion, and spinal function, and perhaps has a reduced risk of adjacent segment degeneration and other spinal de-compensation problems [11]. This method has been shown to retard growth on the convex side while allowing the concave side to continue growing, enabling the abnormal curve to “self-correct” [12]. Additionally, stapling between two adjacent vertebrae at the side of curve convexity inhibits IVD expansion due to its asymmetrical water distribution and therefore, one significant factor for curve progression is eliminated. Theoretically, by stapling the growth plates, the accelerated growth on the convexity of a curve is ceased, both by inhibiting the longitudinal growth (acting directly on the convexwise vertebral end-plate cells) and by eliminating the effect of IVD asymmetrical force application on the growth plates. Perhaps growth is stimulated on the concavity of a curve, and therefore correction of the deformity can occur (Hueter-Volkmann principle). The goal of stapling is to harness the scoliosis patient’s inherent abnormal spinal growth and alter it to achieve correction, rather than progression. 4. Discussion The proposed theoretical model requires three conditions in order to be successful. First, there must be enough growth potential left; otherwise the final correction will be suboptimal. Second, the elastic properties of the IVD should allow adequate modulation and through that adequate alteration of the mechanical environment of the growth plate and third, the corrective forces should be towards the correct level, ideally perpendicular to the level of maximum deformity. The wedging of the elastic IVD in the immature scoliotic spine could be reversed by application of corrective forces on it. Reversal of IVD wedging, by altering the mechanical environment of the adjacent growth plates, is thus amended into a

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T.B. Grivas et al. / The Role of the Intervertebral Disc in Correction of Scoliotic Curves

“corrective”, rather than “progressive”, factor of the deformity. Through the proposed mechanism, treatment of progressive IS with full time and night time braces, exercises and fusionless surgery by anterior stapling could be effective. References [1]

Stokes IA, Analysis and simulation of progressive adolescent scoliosis by biomechanical growth modulation, Eur Spine J 16(10) (2007), 1621-8. [2] Mente PL, Stokes IA, Spence H, Aronsson DD, Progression of vertebral wedging in an asymmetrically loaded rat tail model, Spine 22(12) (1997), 1292-6. [3] Grivas TB, Vasiliadis E, Malakasis M, Mouzakis V, Segos D, Intervertebral disc biomechanics in the pathogenesis of idiopathic scoliosis. Stud Health Technol Inform 123 (2006), 80-3. [4] Taylor T.K.F, Melrose J., The role of the intervertebral disc in adolescent idiopathic scoliosis, Spine: State of the Art Reviews 14 (2000), 359-369. [5] Roberts S., Caterson B., Urban J.B.G, Structure and composition of the cartilage end plate and intervertebral disc in scoliosis, Spine: State of the Art Reviews 14 (2000), 371-381. [6] Melrose J., Gurr K.R., Cole T.C., A. Darvodelsky, Ghosh P., Taylor T.K., The influence of scoliosis and ageing on proteoglycan heterogeneity in the human intervertebral disc, J Orthop Res, 9(1) (1991), 68-77. [7] Bushell G.R., Ghosh P., Taylor T.K., Sutherland, The collagen of the intervertebral disc in adolescent idiopathic scoliosis, J Bone Joint Surg Br, 61(4 J.M.) (1979), 501-8. [8] Dangerfield P.H., Roberts N., Walker J. et al, Investigation of the diurnal variation in the water content of the intervertebral disc using MRI and its implication for scoliosis. In: M. D'Amico, A. Merolli, G. C. Santambrogio (editors). Three Dimensional Analysis of Spinal Deformities. IOS Press 1995, 447-451. [9] Beauchamp M, Labelle H, Grimard G, Stanciu C, Poitras B, Dansereau J, Diurnal variation of Cobb angle measurement in adolescent idiopathic scoliosis. Spine 18(12) (1993), 1581-3. [10] Grivas TB, Rodopoulos GI, Bardakos NV, Biomechanical and Clinical Perspectives on Nighttime Bracing for Adolescent Idiopathic Scoliosis. Stud Health Technol Inform 135 (2008), 274-290. [11] Betz RR, Kim J, D’Andrea LP, Mulcahey MJ, Balsara RK, Clements DH, An Innovative Technique of Vertebral Body Stapling for the Treatment of Patients With Adolescent Idiopathic Scoliosis: A Feasibility, Safety, and Utility Study. Spine 28(20S) (2003), S255–S265. [12] Braun JT, Ogilvie WJ, Akyuz E, Brodke DS, Bachus KN, Fusionless Scoliosis Correction Using a Shape Memory Alloy Staple in the Anterior Thoracic Spine of the Immature Goat. Spine 29(18) (2004), 1980–1989.

Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved. doi:10.3233/978-1-58603-888-5-37

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Ultrasound femoral anteversion (FAV) relative to tibial torsion (TT) is abnormal after school screening for adolescent idiopathic scoliosis (AIS): evaluation by two methods RG BURWELL1, RK AUJLA1, AS KIRBY1, A MOULTON2, PH DANGERFIELD3,BJC FREEMAN4, AA COLE1, FJ POLAK1, RK PRATT1, J K WEBB1 1 Centre for Spinal Studies and Surgery, Nottingham University Hospitals Trust, Queen’s Medical Centre Campus, Nottingham, UK, 2Department of Orthopaedic Surgery, King’s Mill Hospital, Mansfield, UK. 3Children’s Hospital, University of Liverpool, 4Department of Spinal Surgery, Royal Adelaide Hospital, Adelaide, Australia (Supported by AO and Arthritis and Rheumatism Council UK) Abstract. In the scoliotic spine, torsion is generally evaluated in relation to axial rotation of the apical vertebra. In the lower limbs, the changes in torsion by age of femoral anteversion (FAV) relative to tibial torsion (TT) have been studied in dried bones, normal growing subjects and adults and subjects with osteoarthritis of the hip or the knee. This paper reports the application of real-time ultrasound to FAV and TT in normal children age 11-18 years and in scoliosis screening referrals with particular reference to how FAV relates to TT as 1) ratios, and 2) tibiofemoral index (TFI) of torsion, calculated as TT minus femoral FAV. The FAV/TT ratio findings show an abnormal normal relationship of FAV to TT both proximo-distally and in leftright asymmetry. These may express torsional abnormalities in femoral and/or tibial growth plates with left-right asynchrony suggesting the possibility of similar torsional abnormalities in vertebral end-plates and/or rib growth plates initiating the deformity of AIS. TFI of the right limb in the scoliosis girls is greater than in the normals that is interpreted as resulting from earlier skeletal maturation of FAV. FAV/TT ratios and TFI are unrelated to the spinal deformity (Cobb angle and apical vertebral rotation) except for boys where TFI is associated with apical vertebral rotation. FAV/TT ratios may be a more accurate method estimating the relationship of FAV to TT. than TFIs. Key words. Idiopathic scoliosis, pathogenesis, spine, ultrasound, femoral anteversion, tibial torsion, growth plate

1. Introduction 1.1 Torsion of femur relative to tibia in bones, health and osteoarthritis at hip or knee In dry normal adult human femora and tibiae a weak statistically significant correlation was reported between FAV/TT [1]. In healthy children [2] and healthy adults [3-5] FAV/TT did not correlate significantly and no left-right asymmetry was found. In contrast, in adults with osteoarthritis of the hip [6] or knee [7] FAV and TT correlated significantly. Goutallier et al [8] using a tibio-femoral index (TT minus

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R.G. Burwell et al. / Ultrasound FAV Relative to TT is Abnormal After School Screening for AIS

FAV) in lateral or medial knee arthrosis, concluded that femoral and tibial torsions play a part in lateralized knee arthrosis together with frontal plane mechanical factors. There may be ethnic and sex differences in the relationship between torsions of the lower limbs and knee osteoarthrosis [9,10]. The above findings, considered in the perspective of torsions in the normal spine and lower limb bones (Figure 1), suggested the need to evaluate the relation of FAV to TT in girls with AIS. Two methods are used here: 1) FAV/TT ratio, and 2) tibio-femoral index [8]. No data on TFIs for normal subjects are published. 1.2. Normal postnatal changes in femoral and tibial torsion 1.2.1. Femoral anteversion (FAV), or torsion, decreases postnatally into puberty [1114](Figure 1). This age-related change is generally attributed to muscular forces acting across the hip [15], and other mechanical forces [16], but there is a view that external torsion at the distal femoral growth plate (in the same direction as tibial external torsion) also contributes to femoral detorsion [17]. Estrogen may abet mechanical forces to drive femoral detorsion during postnatal growth [13].

Figure 1. Diagram to show the two types of postnatal growth in femora and tibiae – growth in length mainly from growth plates at the knees, and torsion in both bones, in the femur internal torsion (detorsion) through puberty and in the tibia external torsion mainly before puberty. GP=growth plate.

1.2.2. Tibial torsion (TT) increases mainly in the first 5 years of life [18, but see 19] and mostly before puberty [13] aligning the feet in gait before the adolescent growth spurt. TT is thought to be determined at the ankle with the medial malleoli rotating forwards in relation to the lateral malleoli [18,20]. Both FAV and TT express growthplate function influenced partly by mechanical factors and strongly by genetic control [21].

R.G. Burwell et al. / Ultrasound FAV Relative to TT is Abnormal After School Screening for AIS

39

2. Femoral anteversion/tibial torsion correlations are significant, abnormal and asymmetric in the scoliosis subjects 2.1. The scoliosis referrals and normals The scoliosis referrals, age 11-18 years, were referred to hospital by school nurses in routine prescriptive screening for the rotational back shape asymmetry of AIS using a quantitative protocol [22] during 1993-99 (girls 93, boys 35). Clinical evaluation (RGB) identified types of AIS: thoracic (30), thoracolumbar (38), lumbar (25), double (14), pelvic tilt scoliosis (16) and straight spine (5). Normal subjects, age 11-18 years were 301 (girls 157, boys 144) examined in the community by two radiographers (RKA, ASK). The spinal radiograph measurements made by one observer (RGB) included Cobb angle and apical vertebral rotation [23]. 2,2. Ultrasound methods Real-time ultrasound was used to measure femoral anteversion (FAV) and tibial torsion (TT) in the scoliosis referrals and normals by a radiographer (RKA or ASK): FAV was measured between upper end of femur and femoral condyles by the method of Zarate et al [24], and TT between upper tibial shaft and talus by the method of Kirby et al [25]. 2.3. Results A preliminary account of the findings has been published [26]. 2.3.1 Reproducibility The interobserver error (RKA and ASK, n=107, normal subjects) are for FAV: coefficient of reliability left leg 0.82, right leg 0.85 [27]: technical error of measurement left leg 3.1 degrees, right leg 3.0 degrees; intra-class correlation coefficient left leg 0.76, right leg 0.77. For TT: coefficient of reliability left leg 0.85, right leg 0.80: technical error of measurement left leg 4.3 degrees, right leg 4.8 degrees; intra-class correlation coefficient left leg 0.76, right leg 0.74. None of the FAV/TT correlation coefficients for data from normal subjects by the two radiographers are statistically significant. Normal

Scoliosis

Figure 2.Diagram showing the correlations of right and left FAV to TT (FAV/TT) to be weakly significant in normals and very significant in the scoliosis subjects. p-values are shown (***=p<0.001).

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R.G. Burwell et al. / Ultrasound FAV Relative to TT is Abnormal After School Screening for AIS

2.3.2. Normal and scoliosis subjects In the normals, FAV and TT show a weakly significant positive correlation in each leg (right p=0.013, left p=0.014) without asymmetry (p=0.454)(ANOVA after correcting for age and sex). In scoliosis referrals, FAV and TT correlate positively and significantly in each leg (p=0.001) with asymmetry (p=0.009), and very significantly different from normals (right p<0.001, left p=0.008). The FAV/TT correlation in each leg is not significantly different by scoliosis curve side or severity (ANOVA). FAV, TT, left-right differences; and FAV/TT sums are not significantly associated with spinal curve severity (Cobb angle, apical vertebral rotation). 2.4. Conclusion In the scoliosis subjects, the abnormal FAV/TT correlations are evidently determined in femoral and/or tibial growth plates. These FAV/TT findings show an abnormality of the normal relationship of FAV to TT both proximo-distally and in left-right asymmetry. These may express torsional abnormalities in femoral and/or tibial growth plates suggesting the possibility of similar torsional abnormalities in vertebral endplates and/or rib growth plates initiating the deformity of AIS. 3. Tibio-femoral index (TFI) of torsion in normal subjects increase with age but not in girls screened for scoliosis suggesting earlier skeletal maturation 3.1. Definition Tibio-femoral index (TFI) of torsion, calculated as tibial torsion (TT) minus femoral anteversion (FAV)[8]. A preliminary account of the findings has been published [28]. 3,2. Methods and subjects The subjects, screened scoliosis and normals were the same as for the FAV evaluation. FAV and TT were measured as stated above. 3.3. Results 3.3.1 Reproducibility The interobserver error (RKA and ASK, n=88, normal subjects) for TFI is: coefficient of reliability left leg 0.84, right leg 0.78 [26]: technical error of measurement left leg 5 degrees, right leg 6.0 degrees; intra-class correlation coefficient left leg 0.76, right leg 0.70. 3.3.2. Normal children. Normal subjects show an increase in TFI with age (quadratic regression, <0.001, r values: girls right 0.360, left 0.471), boys right 0.515, left 0.504, while from 11-18 years, TFIs on the right increase with age (r & p values: girls right 0.174 p=0.029, left 0.098 NS; boys right r=0.249 p=0.003, left 0.108 NS). In girl scoliosis referrals, TFIs do not increase with age but do so in boys (girls right r=0.014 NS, left r=0.022 NS, boys right r=0.743, p<0.001; left r=0.341 p=0.045) with side differences in boys (p<0.002)

R.G. Burwell et al. / Ultrasound FAV Relative to TT is Abnormal After School Screening for AIS

41

Figure 3. Right leg of normal and scoliosis girls. TFIs plotted against age. The findings for the scoliosis girls are significantly different from the normals (p=0.001) but not for the left leg TFI (p=0.107, ANOVA correcting for age).

3.3.3. Scoliosis referrals, In girl scoliosis referrals, TFIs are 1) in the right leg are significantly different from normal (p right=0.001, left p=0.107, ANOVA corrected for age) and not significant in the boys, and 2) unrelated to curve severity but in boys are associated with apical vertebral rotation but not Cobb angle. 3,4. Conclusions 3.4.1 Normals girls and boys. TFIs increase during growth as TT increases and FAV decreases. 3.4.2 Scoliosis girls In the scoliosis girls, TFIs in the right leg are significantly greater than normal without detectable left-right asymmetry. The greater right TFIs is explained in the scoliosis girls by FAV being significantly less than in normals [14,29] whereas TT is not significantly different from normal [14,30]. This FAV decrease [14.29] may result from abnormally increased femoral detorsion, maturationally earlier than normals [14,29]. FAV asymmetry [14,29] detected by both FAV and FAV/TT ratios is not evident in the TFIs, perhaps because TFI has a greater measurement error than that of FAVs. FAV/TT ratios may be a more accurate method for estimating the relationship of FAV to TT than are TFIs. References [1] [2] [3] [4] [5]

Kobylianski E, WEissman SL, Nathan H, Femoral and tibial torsion: a correlation study in dry bones. Int Orthop 1979, 3(2):145-147 Pasciak M, Stoll TM, Hefti F, Relation of femoral to tibial torsion in children measured by ultrasound. J Pediatr Orthop B 1996, 5(4):268-272. Reikeras O, Is there a relationship between femoral anteversion and leg torsion? Skel Radiol 1991, 20:409-411. Strecker W, Keppier P, Gebhard F et al, Length and torsion of the lower limb. J Bone Joint Surg (Br) 1997, 79-B(6):1019-1023. Seber S, Hazer B, Kose N et al, Rotational profile of the lower extremity and foot progression angle: computerized tomographic examination of 50 male subjects. Arch Orthop Traum Surg 2000, 120(56):255-258.

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[6]

Rittmeister M, Hanusek S, Starker M, Does tibial rotation correlate with femoral anteversion: Implications for hip arthroplasty. J Arthroplasty 2006, 21(4):53-558. et al 2006 Takai S, Sakakida K, Yamashita F et al, Rotational alignment of the lower limb in osteoarthritis of the knee. Int Orthop 1985, 9(3):209-215. Goutallier D, Garabedian JM, Allain J et al, Effect of osseous torsions of the lower limb on the development of lateral femoro-tibial knee arthrosis. Rev Chir Orthop Reparatrice Appar Mot 1997, 83(7):613-621 (French). Tamari K, Tinley P, Briffa A et al, Ethnic- gender-, and age-related differences in femorotibial angle, femoral antetorsion, and tibiofibular torsion: cross-sectional study among healthy Japanese and Australian Caucasians. Clin Anat 2006, 19(1):59-67. Tamari K, Briffa NK, Tinley P et al, Variations in torsion of the lower limb in Japanese and Caucasians with and without knee osteoarthritis. J Rheumatol 2007, 34(1):134-150. Upadhyay SS, Burwell RG, Moulton A et al, Femoral anteversion in healthy children. Application of a new method using ultrasound. J Anat 1990, 169:49-61. Kirby AS, Aujla RK, Burwell RG et al, Torsion in the limb bones of healthy subjects. In Research into Spinal Deformities 1, Studies in Health Technology and Informatics Volume 37, Edited by Sevastik JA and Diab KM, Amsterdam, IOS Press; 1997, 53-56. Burwell RG, Aujla RK, Kirby AS et al, Tibial torsion increase occurs mainly before puberty and femoral anteversion decrease mainly from puberty in normal subjects: An ultrasound study suggests intrinsic (genetic) torsion-sensitivity to estrogen. Clin Anat 21(2):195. Burwell RG, Aujla RK, Kirby AS, Ultrasound femoral anteversion (FAV) and tibial torsion (TT) after school screening for adolescent idiopathic scoliosis (AIS). This Meeting Morscher E, Development and clinical significance of the anteversion of the femoral neck. Reconstr Surg Traumatol 1964, 64:469-472. Tayton E, Femoral anteversion: a necessary angle or an evolutionary vestige?. J Bone Joint Surg (Br) 2007, 89(10):1283-1288. Lau DPC, Femoral anteversion and the nature of the twist in the femur with views on the mechanism of postnatal femoral detorsion. The effects of different lower end datum lines and variations in condylar anatomy in then anteversion angle. A quantitative study. Volumes I & II, B Med Sci Thesis, School of Biomedical Sciences, University of Nottingham, UK, 1987 Kristiansen LP, Gunderson RB, Steen H et al, The normal development of tibial torsion. Skel Radiol 2001, 30(9):519.522. Campos FF, Maiques IAP, The development of tibio-fibular torsion. Surg Radiol Anat 1990, 12:109112. Krishna M, Evans R, Spring A et al, TT measured by ultrasound in children with talipes equinovarus. J Bone Joint Surg 1991, 63-B(2):207-210. Burwell RG, Aujla RK, Kirby AS et al, Percentage changes of tibial torsion and femoral anteversion from a juvenile baseline in normal subjects by age, bone and sex: An ultrasound study suggests mechanisms intrinsic (genetic) to growth plates. Clin Anat 2008, 21(2):195. Burwell RG, Patterson JF, Webb JK et al, School screening for scoliosis – The multiple ATI system of back shape appraisal using the Scoliometer with observations on the sagittal declive angle. In: Surface Topography and Body Deformity. Proceedings of the 5th International Symposium, September 29October 1, Wien 1988, 17-23. Perdriolle R, La Scoliose, son etude tridimensionelle, Maloine S A Editeur, 27 Rue de l’Ecole-deMedicine, 75006, Paris. Zarate R, Cuny C, Sazos P, Determination de l’anteversion du col du femur par echographie. J de Radiol 1983, 64:307-311. Kirby AS, Burwell RG, Aujla RK et al, A new method for measuring tibial torsion (TT) angles and findings for young adolescents. Clin Anat 1994, 7(1):55-56. Burwell RG, Aujla RK, Kirby AS et al, Ultrasound femoral anteversion/tibial torsion correlations are significant, abnormal and asymmetric after school screening for adolescent idiopathic scoliosis (AIS): lower limb torsional markers for initiation of the trunk torsional deformity of AIS?. Clin Anat 2007, 20(7):855. Ulijasek SJ, Mascie-Taylor CGN, Chapter 3, Intra- and inter-observer error in anthropometric measurement. In Anthropometry: the individual and the population. Cambridge:University Press, 1994, pp 30-55. Burwell RG, Aujla RK, Kirby AS et al, Tibio-femoral index (TFI) of torsion: an increase with age in normal subjects but not in girls screened for scoliosis suggests earlier skeletal maturation: an ultrasound study. Clin Anat 2008, 21(2):195.

[7] [8]

[9]

[10] [11] [12]

[13]

[14] [15] [16] [17]

[18] [19] [20] [21]

[22]

[23] [24] [25] [26]

[27]

[28]

R.G. Burwell et al. / Ultrasound FAV Relative to TT is Abnormal After School Screening for AIS

43

[29] Burwell RG, Aujla RK, Kirby AS et al, Ultrasound femoral anteversion (FAV) is decreased and asymmetric after school screening for adolescent idiopathic scoliosis (AIS): femora show torsional anomalies that if in the trunk may initiate the deformity [abstract]. Clin Anat 2007, 20(7):855. [30] Burwell RG, Aujla RK, Kirby AS et al, Ultrasound tibial torsion (TT) and TT asymmetry are not abnormal after school screening for adolescent idiopathic scoliosis (AIS): in scoliosis boys TT is decreased relative to scoliosis girls without asymmetry - the result of altered maturation at knee tibial growth plates? Clin Anat 2007, 20(7):855.

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Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved. doi:10.3233/978-1-58603-888-5-44

Risser sign: the value of the lateral spinal radiograph to assess the excursion of the iliac apophysis T KOTWICKI Department of Pediatric Orthopedics and Traumatology University of Medical Sciences, Poznan, Poland [email protected] Abstract. The course of the ossification of the iliac apophysis is considered in adolescent patients with idiopathic scoliosis, under the name of the Risser sign, to determine the remaining spinal growth. Although the iliac crest develops in the three-dimensional space as a complex structure, the iliac apophysis ossification has been assessed only on a one plane frontal spinal radiograph. This study points out the usefulness of the lateral radiograph for the visualization of the whole iliac crest, especially the posterior region which otherwise cannot be observed. Two young female pelvis specimen were examined with anatomical measurements and radiography. Lateral spinal radiographs of 201 girls were analyzed for the iliac apophysis excursion. The measures of the width of the iliac bone beneath the iliac crest revealed one anterior and one posterior thick regions, coupled with an intermediate thin region. The regions of the maximal thickness corresponded to the earliest appearance of the apophysis ossification (Risser 1), while the thin part of the iliac bone corresponded to late appearance of the apophysis ossification (Risser 3-4). The ossification of the posterior part of the crest was best visualized with the lateral radiograph, which was exclusive in showing the posterior superior iliac spine region. On the frontal spinal radiograph the end of the course of the apophysis (Risser 3-4) is usually searched at the level of the sacroiliac joint, while in reality this point was found to be situated more caudal, and accessible for observation on the lateral radiograph.

1.

Introduction

The cranio-caudal growth of the human spine is not linear; the main phases of the most intensive growth concern the first two years of age as well as the early puberty (usually 11-13 years in girls). Increase of the structural scoliotic curvature occurs during this rapid growth phase [1, 2]. The growth potential running out, the evolution of a spinal deviation loses in its dynamics. Thus, the knowledge of the developmental stage of an adolescent with scoliosis is crucial for making therapeutic decisions: planning of surgery, brace treatment or brace weaning. In adolescence the biological age does not always correspond to the chronological age. That is why a number of indices has been developed to assess the bone age with radiography. The Risser sign [3] is one of the most popular. The determination of the Risser sign value (from zero to five) reposes on the assessment of the excursion of the secondary ossification center of the iliac crest (iliac apophysis). In is believed that the ossification starts at the superior anterior iliac spine (SAIS), continues along the iliac crest from anterior to posterior and completes at the posterior superior iliac spine (PSIS). Minor asymmetries of the left-right apophysis

T. Kotwicki / Risser Sign

45

ossification have been described [4]. The assessment of the Risser sign value is performed after an antero-posterior (AP) or a postero-anterior (PA) spinal radiograph comprising the pelvis. The assessment after the lateral spinal radiograph has not been reported. The complex shape of the female pelvis reflects the function of the osseous pelvic girdle providing stability for transmitting the trunk load to the lower limbs throughout the pelvic ring. The most upper part of the left and right iliac bones, namely the iliac crests develop their shape in the three dimensional (3-D) space. Starting from the ASIS the iliac crest continues in the superior and posterior direction, then posterior, then progressively more medial to the L5 level and finally rapidly down and posterior up to the PSIS. As any 3-D structure cannot be seen on one plane radiograph, it is admitted in orthopedic surgery to systematically visualize bony structures using two perpendicular projections: a frontal one and a lateral (or axial) one. However, only the frontal one has been used so far to evaluate the iliac apophysis excursion for the Risser sign value. The aim of the study was to investigate the size, shape, width and the spatial orientation of the iliac crest using two anatomic specimen and to assess the development of the iliac apophysis using lateral radiography.

Fig. 1. The pelvic specimen (bottom) and its radiography (top), both seen in the frontal (left), transverse (middle) and lateral (right) plane. ASIS – anterior superior iliac spine. PSIS – posterior superior iliac spine. aPSIS – apparent PSIS – the point commonly considered to be the PSIS, which is actually situated at the junction of the 70% anterior and the 30% posterior of the total length of the iliac crest. This posterior part can be seen on the lateral radiograph exclusively.

2.

Material and Methods

Two young female pelvic specimen were examined with frontal and lateral radiography, attaching metal markers along the iliac crest (Fig. 1). The thickness of the iliac bone was measured just beneath the iliac crest, along the whole distance from the ASIS to the PSIS at 1cm intervals. Moreover lateral radiographs of 201 girls with

46

T. Kotwicki / Risser Sign

idiopathic scoliosis, aged 10.2 to 20.0 years, mean 14.6 ± 2.2 years, were examined to reveal the excursion of the apophysis. The X-rays were taken from the charts of the patients, and not ordered because of this study. The position of the PSIS was studied and related to the vertebral level (to the vertebral body level or the intervertebral disc level). 3.

Results

The length of the iliac crest of was 23.5cm for the first specimen, and 21.5cm for the second one. The iliac bone was the thickest in two regions, one adjacent to the ASIS and another one to the PSIS. In the ASIS region the width of the iliac bone varied from 10.7mm to 17.3mm, mean 13.7mm (the first specimen), and from 11.2mm to 14.9mm, mean 13.1mm (the second specimen). In the region of the PSIS the width of the iliac bone varied from 10.4mm to 15.7mm, mean 12.8mm (the first specimen), and from 11.4mm to 17.6mm, mean 15.5mm (the second one). The iliac bone was thin in between the two above mentioned regions. In the first specimen the width of the thin region varied from 5.8mm to 7.4mm, mean 6.5mm, while in the second specimen the width varied from 8.1mm to 10.1mm, mean 9.6mm. The radiography of the pelvic specimen revealed that the regions of the maximal width of the iliac bone corresponded to the regions of the first appearance of the ossification of the iliac crest. The analysis of the position of the PSIS revealed the level of L5 in 3 cases, the disc L5/S1 in 4 cases, the S1 in 96 cases, the disc S1/S2 in 52 cases and S2 in 35 cases. The ossification of the iliac apophysis was clearly seen on the lateral radiograph. Various stages of the ossification were observed: (1) no ossification, (2) short and thin shadows of the beginning ossification, (3) full length excursion or (4) fusion of the apophysis with the iliac crest (Fig. 2.). Thus, the lateral radiograph excursion of the iliac apophysis was proposed to be quantified in four grades [paper submitted for publication].

Fig. 2. Lateral radiograph showing the full excursion of the iliac apophysis up to the posterior superior iliac spine. The most posterior and inferior portion of the iliac crest is not visible on the frontal plane radiograph, however it can be observed on the lateral one.

T. Kotwicki / Risser Sign

4.

47

Discussion

This study confirmed what is already known from anatomical studies, that the value of the width of the iliac bone can present important variations, and that the regions of maximal or minimal width are distributed in the systematic way: the anterior maximum, the middle minimum, the posterior maximum. The knowledge of the size of the iliac bone is useful in clinical practice: both the anterior and the posterior thick regions are the preferable sources to harvest the bone graft in orthopedic surgery. It was interesting to notice that the regions of the iliac bone which revealed the maximal width corresponded to the regions of the first appearance of the apophysis ossification (Risser 1). The amount of the underlining bone would be the main reason for early appearance of the ossification in particular regions of the iliac crest. Also, the sequence typical for the Risser sign development may be explained by the sequence of ossification beginning at the large bony regions and followed by the progressive ossification of the thinner regions. It worth mentioning that the region of the posterior maximum thickness of the iliac bone cannot be seen on the frontal pelvic radiograph. On the other hand, it is well seen on the lateral radiograph. When studying the representations of the Risser sign which are reproduced in the manuals of orthopedics, one can easily notice that the excursion of the posterior apophysis is designed as stopping at the level of L5 vertebra, lateral and superior to the sacroiliac joint. In this study the PSIS was found to be situated much lower, at the level of S1 to S2 vertebra. These findings strongly support the thesis that there exists a posterior part of the iliac crest which cannot be observed on the frontal radiograph, however it can be seen on the lateral one. 5.

Conclusions

The complex 3-D shape of the iliac bone bordered with the iliac crest cannot be accurately studied from the one plane frontal radiograph. The additional use of the lateral radiograph allows detecting of the ossification of the posterior part of the iliac crest. The thickness of the iliac bone represents two maxima joined with a thin region in the middle. The thick regions correspond to the radiographic early appearance of the ossification of the iliac apophysis (Risser 1) while the thin regions correspond to the late ossification (Risser 3-4). References [1] Bunnell WP. The natural history of idiopathic scoliosis before skeletal maturity. Spine 1986; 11: 773-776. [2] Lonstein J Carlson JM. The prediction of curve progression in untreated idiopathic scoliosis during growth. J Bone Joint Surg Am. 1984; 66: 1061-1071. [3] Risser JC. The iliac apophysis. An invaluable sign in the management of scoliosis. Clin Orthop 1958; 11: 111-119. [4] Little DG Sussman M. The Risser sign: a critical analysis. J Pediatr Orthop 1994; 14: 569-575.

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Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved. doi:10.3233/978-1-58603-888-5-48

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Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved. doi:10.3233/978-1-58603-888-5-52

Clinical detectable tension in the growing body: new and revisited signs in clinical examination in children with postural problems and spinal deformities Restoration of lordosis on the thoracolumbar junction can correct sagittal and coronal plane deformity; a new (revisited) linked approach on the treatment and etiology of adolescent spinal deformities

P J.M. van Loon, MD. Department of orthopaedic surgery 1 Slingeland Hospital, Doetinchem, The Netherlands Abstract: Unclear etiology in scoliotic and kyphotic deformities of the spine is responsible for uncertainty in treatment options. Normal all-day factors can be of importance. Newly developed or revisited clinical examination of sitting and supine children and consequent testing of neuro-muscular tightness shows to be useful in understanding the different spinal deformations and postural problems during growth and point to neuromuscular tension in growth. The goal is: -Better understanding of the role and individual characteristics of the central nervous system, especially the cord and roots in proper and improper growth of the human spine.- Clarifying that preservation of lordosis and good function at the thoracolumbar junction at the end of growth can be of value for normal configuration and function of the spine in adult life.Present obvious important and consistent clinical observations in children in sitting and supine position with early and advanced adolescent deformities, by photographic studies and video fragments. --Use of work on growth and deformation of the spine by Milan Roth on uncoupled neuro-osseous growth and other historical literature. -Relate these clinical findings and background literature with common knowledge about adolescent spinal deformities and mechanical laws on tensile and compressive forces in structures. Overview of relevant clinical tests in the growing child presented with deformities show possible correlation with the proposed internal balancing problem (uncoupled neuro- osseous growth) researched by Roth. Concomitant radiological and MRI signs are shown. Around 1900 most orthopaedic surgeons and anatomists saw relationship between the new habitude of children to sit for prolonged periods in schools and spinal deformities. A physiological explanation as adaptations needed by the total neuromuscular system (“the growing system”) was widely postulated ( Hueter-Volkmannprinciple) and subject in research but a concise theory was not achieved. By recognising positive effects of creating lordosis at the thoracolumbar junction of the spine and consistent clinical findings in early deformations scientific support was found by earlier experimental work of Roth. With a leading role of the central nervous system in growth of the spine of standing and sitting vertebrates by steering a tension based system, deformation can be understand as adaptations. Consequences for new preventive measures and therapeutic strategies in deformities seems possible Keywords: scoliosis, kyphosis, spinal tension, biomechanics, thoracolumbar spine, osteoneural growth, historic studies

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1. Introduction If scoliosis and other deformities are assumed to be the visible results (using X-rays as the gold standard for diagnosis) of a mismatch in the speed and control of growth between the different systems of the body and tensile forces which contribute to these differences, it is important to know if these tensile forces (or tensions) are detectable is growing children. If so, new tools in assessment, screening and treatment can be developed. Tension is always present in the musculo-skeletal system. No posture or movement is possible without a certain amount of this tension developing. Roth’s spring models of deformities (Fig. 1 and 2) are ways of understanding the links between these physical forces relating to the complex spine/cord relationships. This paper reviews aspects of the work of Roth in this area, developed in conjunction with his practice devoted to examining children, and highlighting his contribution to scoliosis research, with the object of stimulating debate on his ideas and contributions [1-6]. In 1927, Lambrinudi’s work on scoliosis noted the importance of assessing muscular tightness in the legs in relationship to a progressing spinal deformity [7, 8]. In recent years, I have revisited and incorporated several tests on neuromuscular tightness and the assessing of functional properties of the spine into the clinical investigation of children. Photographs and video-fragments will be used in the lecture to demonstrate this. The tests for tensile forces give clues on factors leading to progression of deformities. These are: 1.) The use of the Adam’s bending test from the posterior (torsion) and the lateral positions; the type of kyphotic curvature present and the likelihood of achieving a horizontal lumbar spine; 2.) A “scoop” test on thoracolumbar extension pattern, using the head coming up first from the bending test with the neck grasped by both hands. A normal spine firstly creates a “scoop” or lordosis at the TL area before the hips extend. Early deformities present as a kyphosis while the hips are extended. 3) A straight leg sitting test on spinal mobility: with tense neuromuscular structures, children without visible deformation in the standing position will sit more on their sacrum than on the ischial tuberosities and will thus not be able to stretch or even lordose their spines. Sitting the subject on the edge of an examination couch gives space for the long lever-arms of the upper legs and restores opportunities for stretching and lordosing the spine. 4.) The Unilateral Straight Leg Raising test in supine children: assessment of the leg-couch angle and/or femorotibial angle: This is widely used in Scheuermann’s disease [9] and is often found different between the left and right sides in scoliosis. In the early progressing phase of scoliosis some real tightness will sometimes be detected. 5.) Bilateral Straight Leg Raising test: tightness will be found when raising the feet in a fixed stretched position of the hips and knees, followed by lifting of the buttocks off the couch, extending up into the whole spine as far as the shoulders. (This is referred to “Brettsteiff” in German ie as stiff as a board). 6.) A ‘Jack in the box’ reflex test: assessment of the protecting tightness while lowering the lifted legs back onto the couch after bilateral straight leg raising. In children with a tense trunk, they lift their head or involuntary assume a more than complete sitting position.

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The variability found in all these tests is assumed to be dependant on multiple factors like age, gender, joint mobility, familiar predisposition, “stiffness” in gymnastics, phase of growth spurts, results of bodily exercises, good compliance in bracing etc. It makes scientific, reproducible measurements less reliable than the gold standard used for measurements on X-rays and has had to be interpreted on an individual basis. An important issue is the visualisation and recognition of the children and their parents of the sometimes unusual findings in these tests. Their subsequent disappearence as a result of good exercise or brace compliance offers a positive psychological effect in normally long-term treatment. Where do these tests fits in the neurovertebral and osteoneural growth relations? As discussed elsewhere in this volume in the context of the contributions of Milan Roth to understanding scoliosis, his views on the causation of scoliosis state that: “Idiopathic scoliosis may be interpreted as an adaptive morphogenetic reaction of the vertebral column upon the growth insufficiency of the intraspinal nervous structures: The growth process of the vertebral column, though continuing undisturbed at the cellular level, is adapted at the organ level by “waves” to the growth-insufficient cordnerve complex with the musculature as the force delivering organ. Morphological features of the scoliotic vertebra together with the typical position of the spinal cord within the spinal canal speak in favour of the suggested vertebro-neural concept which offers a plausible explanation of the congenital and experimental scoliosis as well”.

Figure1 Roth’s simple model using a spring and a thread to represent a scoliotic deformity demonstrate where tensile and compressive forces are located.

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Figure 2 Roth’s simple model using a spring and a thread to represent a kyphotic deformity demonstrate where tensile and compressive forces are located. As in figure 1 changing terms as lordosis or scoliosis into unilateral compression and contra lateral distraction or shortening and lengthening helps in understanding the role of tension as a present force.

Roth is equally clear about what happens in adjusting growing forces, and the therapeutic implications this might have: “Adjustment or rectification of the deformed structure like a scoliotic spine is not accompanied by lengthening, notwithstanding the fact that the scoliotic trunk is elongated; the curved spine is adjusted, not lengthened. The adjustment involves a contraction or reduction of the convexe side of the discs. Consequently in adjusting– paradoxally but irrefutably- the spinal canal is rather shortened”. In our opinion he considered that the natural actions embodied within the HueterVolkmann principle could be reversed by an elastic (tensile) deforming force. The question is now posed relating to how we can achieve modifying the position and curvature of the spine so that these tensile forces and their producing muscles can be addressed. It should be noted that providing the answer to this question was not the principle object of Roth’s research. However, some clues, which might lead to an answer to the problem, can be found elsewhere in Roth’s work. Here, he stated that a lordotic form of the lumbar and thoracolumbar spine is of great importance for proper function of this complex and mobile region of the spine in childhood. This would then facilitate an improved function within the lower thoracic ribs cage, which are elevated by muscular forces and also retro-pulsed themselves. The bellows-like function of the diaphragm is also optimised by these actions. The practical outcomes of this hypothesis will be discussed later. 2. Disproportionate growth Roth paved the way to develop a new framework of neurovertebral and osteoneural growth relations, which would allow research into these concepts of etiology of scoliosis. By introducing the research community to the work of previous generations

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of scientists, like Roth, there is hope to stimulate debate and new directions of research with modern-time possibilities[10-13]. For us, Roth’s findings on disproportionate growth mean that an elegant incorporation of his concepts into modern research becomes possible. By developing these concepts, a new coherent system of interdependent hypotheses for the explanation of growth, and the (mal) functioning of the spine and the cord may be advanced. If form always follows function and no (muscular) function is without purpose, osteoneural growth relations are controlled. In turn, this could give us clues for formulating further research on proper treatment or prevention. How did we get to this statement? The published work of Roth had to be reconciled with other studies of natural behaviour: 1 the very interesting work of Gracovetsky and Farfan on the optimum spine or where nature is heading for; 2 with developments in the last ten years in biomechanics or how does nature do things in order to copy this (robotology, prosthesiology), 3 with own clinical observations on tension related features or how nature shows itself For me, the most elegant feature of these three concepts is that of the connecting element or type of forces that connects all of these three concept lines This is tension in its different manifestations and this, natural, element that is able to withstand gravity should be incorporated into orthopaedic biomechanical knowledge 3. The elements of the new concepts The formulation of the main elements of this system of concepts fall into three parts. 3.1 Roth’s concept of disproportionate growth of the cord vs. the skeleton. Roth’s concept of neurovertebral and osteoneural growth relations of the cord vs. the skeleton(and joints and discs) implies that there should be a clear and challenging domain for what triggers the manifestation of several spinal deformities. It means that research in this field can then be directed from nowadays interest in bone growth and hormonal pathways towards the causes of differences in the quality of cord stretching and the intrinsic role of growth spurts in that type of growth. While this is not the normal domain for orthopaedic surgeons, it does set a clear agenda for co-operation with geneticists, neurologists, neurosurgeons and paediatricians to research these concepts further. The way disproportionate growth manifests itself in a (de) formation of the spine however is another separate subject and field of research. There clinical tests and practical possibilities to intervene (exercises, bracing) to stop progression or correct already present deformities come into the practice of orthopaedics. From this presumed cause of conflicting elements in two types of growth in one body that works out in the actual formation of the spine then divides in two different concepts. 3.2 How normal movements and posture create the shape of the spine. In this area, we feel very comfortable with the progress in biomechanics that has been made in the last ten years. New ideas and concepts have been developed particularly by Gracovetsky and Farfan [14, 15]. In their work on the optimum spine, but also by

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several other authors [16, 17]. Movement is necessary for proper development of a child’s body. The issue what could be suggested or added in those research lines is getting improved knowledge of characteristics of the thoracolumbar area in a good functioning spine as in pre-adolescents and the role of always present tensile forces in a living body. 3.3 How moulding and shape of the bony elements of the spine occurs. Given the concepts embodied in ideas 1 and 2, how does moulding (in embryologic and postnatal growth) of the bony elements of the spine occur? We consider that, for proper understanding, a “multi-component mortar” concept is required. Bone tissue grows by normal cell divisions as a part of the normal cell cycle. Every bony element is a result of generation (osteoblasts) and degeneration (osteoclasts) of cells where the pace of cell division creates the shape as we see it. It is also known that the resultant pace of growth is different throughout all bony structures. In the concept of “multi component mortar”, tension as an underestimated natural force forms the activating component that then triggers the other biochemical components of the “mortar” to format certain dimensions. This is very well researched in trees with applications in engeneering18, 19 4. Conclusion: the application of the concepts. In practice and through recent research, I have concentrated on applying the concepts under 1 and 2 on how to restore spinal deformities following the principles as stated by Roth. My conclusion is that restoring lordosis through the thoracolumbar joint appears to be the key to achieving the appropriate adjustments to the total, inter-connected components of the spine. The thoracolumbar joint function in what should, or can, be seen as a three-dimensional hinge in a complex, connected inverted pendulum, the spine, strives to maintain an equilibrium state. Where form follows function, the coordinated neuro-muscular tension of the dynamic tension cables, the musculature, directs the positioning and form of the bony structures in order to create an intrinsically well-balanced system. In our recent article in Spine 2008 we presented what might be called a controlled test of the principle of the working of lordotic intervention [20]. We tested the hypothesis that correction of scoliosis may benefit from a lordotic fulcrum force in the sagittal plane on the thoracolumbar spine. The results were statistically significant. Additionally, during the last five years we have developed a spinal correction method based on this principle called thoracolumbar lordotic intervention (TLI). As a matter of fact not yet published on the neuromechanic but only biomechanic results I consequently experienced in the performance of pedicle subtraction osteotomies at the thoracolumbar spine in deformities a impressive tension releasing feature [21]. In TLI-bracing we do not put corrective force on the bone structures, but more logically, onto the tension cables, namely the muscles. Rearrangement of tensile forces is required because of a complete chance in the postural balance. Instead of a slight or clear flexed trunk position, we restore antero-posterior curvatures, and thus, this rearrangement of masses and muscle forces needed to control balance will end in a corrected three-dimensional posture. Then the presence or need of “tight hamstrings” disappears. We gently force the child’s growing body to auto correct the deformity

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while we focus on intervening only in the sagittal plane. Shoulders and buttocks regain their relative posterior position, while the thoracolumbar area is passively and actively placed at a more anterior position. The first results in this type of “conservative” treatment were presented at the SOSORT-Athens 2008 meeting giving strong support to the incorporation of Roth’s work in further therapeutic directions. To revisit older or almost forgotten work of earlier scientists, not only in orthopaedics but other fields, allows the possibilities of linking such concepts and ideas together leading to an innovative path for a range of medical and scientific disciplines. References [1] [2] [3] [4] [5] [6] [7] [8] [9] [10]

[11]

[12]

[13]

[14] [15] [16] [17] [18] [19] [20]

[21]

Roth M. [Some normal and pathologic aspects of growth relations between the spinal cord and the spine in the pneumomyelographic picture]. Cesk Radiol 1967 Jan;21(1):1-12. Roth M. Idiopathic scoliosis caused by a short spinal cord. Acta Radiol Diagn (Stockh) 1968 May;7(3):257-71. Roth M. [Relative osteo-neural growth--some phylogenetic, ontogenetic and clinical aspects]. Radiol Diagn (Berl) 1971;12(1):81-97. Roth M. [Morphogenesis of physiological and developmentally conditioned pathological curvatures of the spine]. Cesk Radiol 1972 Jul;26(4):179-88. Roth M. [Neuromorphology of joint movement]. Cesk Neurol Neurochir 1981 Nov;44(6):341-9. Roth M. [Idiopathic scoliosis and Scheuermann's disease: essentially identical manifestations of neurovertebral growth disproportion]. Radiol Diagn (Berl) 1981;22(3):380-91. Lambrinudi C. A method of treating lumbar scoliosis. Proceedings of the Royal Society of Medicine 1927. Lambrinudi C. Adolescent and senile kyphosis. British Medical Journal 1934;2:800-4. Hosman AJ, de KM, Anderson PG, van LJ, Langeloo DD, Veth RP, et al. Scheuermann kyphosis: the importance of tight hamstrings in the surgical correction. Spine 2003 Oct 1;28(19):2252-9. Chu WC, Lam WW, Chan YL, Ng BK, Lam TP, Lee KM, et al. Relative shortening and functional tethering of spinal cord in adolescent idiopathic scoliosis?: study with multiplanar reformat magnetic resonance imaging and somatosensory evoked potential. Spine 2006 Jan 1;31(1):E19-E25. Chu WC, Ng BK, Li AM, Lam TP, Lam WW, Cheng JC. Dynamic magnetic resonance imaging in assessing lung function in adolescent idiopathic scoliosis: a pilot study of comparison before and after posterior spinal fusion. J Orthop Surg 2007;2:20. Chu WC, Man GC, Lam WW, Yeung BH, Chau WW, Ng BK, et al. A detailed morphologic and functional magnetic resonance imaging study of the craniocervical junction in adolescent idiopathic scoliosis. Spine 2007 Jul 1;32(15):1667-74. Chu WC, Man GC, Lam WW, Yeung BH, Chau WW, Ng BK, et al. Morphological and functional electrophysiological evidence of relative spinal cord tethering in adolescent idiopathic scoliosis. Spine 2008 Mar 15;33(6):673-80. Gracovetsky S, Farfan H. The optimum spine. Spine 1986 Jul;11(6):543-73. Gracovetsky S. An hypothesis for the role of the spine in human locomotion: a challenge to current thinking. J Biomed Eng 1985 Jul;7(3):205-16. Cholewicki J, Panjabi MM, Khachatryan A. Stabilizing function of trunk flexor-extensor muscles around a neutral spine posture. Spine 1997 Oct 1;22(19):2207-12. MacKinnon CD, Winter DA. Control of whole body balance in the frontal plane during human walking. J Biomech 1993 Jun;26(6):633-44. Mattheck Claus. Biomechanical Optimum in Wooden Stems. In: Barbara L.Gartner, editor. Plant Stems: Physiology and Functional Morphology. Academic Press; 1995. Mattheck C, Bethge K, Erb D, Blomer W. Successful shape optimisation of a pedicular screw. Med Biol Eng Comput 1992 Jul;30(4):446-8. van Loon PJ, Kuhbauch BA, Thunnissen FB. Forced lordosis on the thoracolumbar junction can correct coronal plane deformity in adolescents with double major curve pattern idiopathic scoliosis. Spine 2008 Apr 1;33(7):797-801. van Loon PJ, van SG, van Loon CJ, van Susante JL. A pedicle subtraction osteotomy as an adjunctive tool in the surgical treatment of a rigid thoracolumbar hyperkyphosis; a preliminary report. Spine J 2006 Mar;6(2):195-200.

Chapter 3 Biomechanics, Surface Topography and Imaging

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Analysis of the mechanisms of idiopathic scoliosis progression using finite element simulation X.DREVELLE*1, J.DUBOUSSET1, Y.LAFON1, E.EBERMEYER2, W.SKALLI1 1

Laboratoire de Biomécanique, Arts & Métiers ParisTech – CNRS UMR 8005, Paris, France 2 University School of Medicine, Saint-Etienne, France

Abstract. The mechanisms of idiopathic scoliosis progression are still not fully understood. The aim of this study is to explore, using finite element simulation, effect of the combination of gravity and anterior spinal overgrowth on scoliosis progression. 14 adolescents (10 girls, 4 boys) with an average age of 10.8 years [range 9; 13] were divided in three groups: thoracolumbar scoliosis (TL), lumbar scoliosis (L), asymptomatic patients (A). Accurate 3D reconstructions of the spine have been built using bi-planar X-rays. A patient specific validated finite element model has been used. Simulations have been launched with simulation of the combined effect of gravity and growth. The progression during the simulation was defined by a maximal axial rotation movement greater or equal than 4° and a maximal lateral displacement greater or equal than 5 mm (“first order progression” for one criterion, “second order” for the both criteria). In the group TL, we notice an aggravation for 4 patients (Cobb angle increase at least by 4°, mean at 5.9°). Only three patients of the group L show a progression with a smaller Cobb angle increase (mean 3.9°). For the group A, no progression is found for 3 and a progression is found for 1. An anterior spinal overgrowth combined with gravity and a preexistent curve in the spine could lead to a progression of scoliosis. It seems necessary to consider differently lumbar curves from other curves. Numerical simulation with a patient specific model appears as a useful tool to investigate mechanisms of scoliosis aggravation. Keywords: scoliosis; progression; growth; finite element simulation

1.

Introduction

Because of the number of factors involved, the mechanisms of idiopathic scoliosis progression are still not fully understood. These mechanisms are bound both to the nature of the deformation and to the growth process. Particularly the factors that induce the well known torsion phenomenon with vertebral axial rotation, lateral deviation and sagittal extension still remain obscure. The aim of this study is to explore, using finite element simulation, biomechanical mechanisms involved in the progression of scoliosis. We will focus on the combination of gravity and anterior spinal overgrowth. *

Corresponding author. Email: [email protected]

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X. Drevelle et al. / Analysis of the Mechanisms of Idiopathic Scoliosis Progression

Materials and methods

Fourteen adolescents (10 girls, 4 boys) with an average age of 10.8 years [9; 13] were divided in three groups: thoraco-lumbar group (TL), lumbar group (L) and asymptomatic group (A) (Table 1). The group TL is composed of 4 patients (3 girls, 1 boy) who have a thoracolumbar scoliosis, with an average age of 10.3 years [range 9.5; 11] and a mean Cobb angle of 13.3° [range 8.8°; 17°]. The group L includes 6 patients (5 girls, 1 boy) who have a lumbar scoliosis. The patients present an average age of 11.3 years [range 9; 13] and a mean Cobb angle of 11.1°[range 3.1°; 18.8°]. The group A is composed of 4 asymptomatic patients (2 girls, 2 boys). The patients present an average age of 10.3 years [range 9; 12].

The X-Rays data used for the groups TL and L were performed within routine exams after their first visit for scoliosis. All the patients of groups TL and L present a Risser index of 0. Patients of the group A are involved in a parallel project which was led at the Laboratoire de Biomécanique (with ethics committee approval). Using bi-planar X-rays and an accurate reconstruction method [1], accurate 3D reconstructions are obtained (Figure 1.a). A patient specific finite element model, which was described and validated in a previous study [2; 3], is built from the reconstructions (Figure 1.b). For each functional unit, a global model is considered. Vertebrae are represented using stiff elastic beams connecting attachment points for discs and ligaments. Surfaces are attached to these beams to represent joint facets, their orientation being defined according to the vertebral level. Ligaments are modelled using tension-only cable elements. The pelvis is taken into account as a quasi-rigid body represented by a set of stiff beams connecting attachment points of iliolumbar ligaments. Discs are modelled using modified beam elements: to take into account the torsional effects of the disc fibers, mechanical properties are set to differentiate behaviour in bending and torsion.

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All degrees of freedom of each node modelling the pelvis are blocked. The z-axis translation is free and all others degrees of freedom are blocked for T1. The gravity effect is modelled by a 400N z-axis compression force on T1 vertebral body. The anterior spinal overgrowth is modelled by a 15% z-axis lengthening of each vertebral body (i.e. on each beam modelling each vertebral body) with no specific constraints on the posterior part. For each patient a simulation has been launched with the combined effect of gravity and growth. The progression during the simulation is defined by a maximal axial rotation movement greater or equal than 4° and a maximal lateral displacement greater or equal than 5 mm. When one criteria is observed, the progression is classified “P1” and “P2” when both criteria are observed. The observations are completed with the modification of the Cobb angle and the T5-T12 kyphosis. 3.

Results

Simulations show various results for the subjects (Table 2). In the TL group, we notice a progression for 4 patients on 4.The Cobb angle increases at least by 4° (mean 5.9°). The mean maximal movement for axial rotation and lateral displacement are respectively 4.6° [range 3.0°; 7.3°] and 8.0mm [range 5.7mm; 10.0mm]. Considering the L group, 3 patients on 6 show a progression with a smaller Cobb angle increase (mean 3.9°). For these three patients, the mean maximal movement is 4.8° [range 4.7°; 5.0°] for axial rotation and 4.3mm [range 2.7mm; 5.1mm] for lateral displacement. For the other three patients, these values are 2.5° [range 2.0°; 2.5°] for axial rotation and 2.2mm [range 1.8mm; 2.6mm] for lateral displacement. For the group A, maximal movement is under 3° for the axial rotation and under 5mm for the lateral displacement (for all except one). For all the 14 patients, we notice a decrease of the thoracic kyphosis (mean -7.4°, [range -2.8°; -11.9°]). In some cases the thoracic curve became lordotic.

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X. Drevelle et al. / Analysis of the Mechanisms of Idiopathic Scoliosis Progression

Discussion and conclusion

Several authors have investigated the anterior spinal overgrowth leading to scoliosis progression according to the “rotational lordosis” [4; 5].Some have developed this theory by defining the “vicious cycle” [6] based on the asymmetrical growth due to the stress distribution. On this study we focus only on the spinal anterior overgrowth without growth modulation and use finite element simulation to investigate this. Several others studies use numerical simulation to investigate scoliosis progression. Most of them support the “Hueter-Volkmann law” with integration of growth modulation [7; 8]. However some authors have already investigated the “anterior spinal overgrowth by using the finite element simulation. Azegami et al investigate the reaction of the spine under different buckling mode inducing by thoracic anterior spinal overgrowth [9]. Goto et al investigate the influence of bone modelling on the same buckling mode [10]. Both found that the second bending mode on the sagittal plane due to thoracic anterior spinal overgrowth combined with bone resorption lead to scoliosis and progression. The results of this preliminary study support this theory. It seems that with some pre-existing deformations anterior growth can explain lateral deviation, axial rotation and Cobb angle progression. This Cobb angle increase could itself yield an asymmetric growth as a secondary effect. However this combination seems not to be an initiating factor on a normal spine, while it could aggravate a pre-existing deformation. Even if the number of subjects is too small to perform statistical tests, initial pattern plays a particular role: TL group seem more likely to progress under our hypothesis than L group. Then, the mechanisms of progression seem to be different in lumbar curves and in thoraco-lumbar ones. An investigation on a larger group of subjects will provide us a better understanding of scoliosis pattern role. Numerical simulation with a patient specific model appears as a useful tool to investigate mechanisms of scoliosis aggravation. • An anterior spinal overgrowth combined with gravity and a pre-existent curve in the spine could lead to scoliosis progression. • It seems necessary to consider differently lumbar curves progression mechanisms from other curves. • Numerical simulation with a patient specific model appears as a useful tool to investigate mechanisms of scoliosis aggravation.

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Acknowledgments The authors gratefully acknowledge S.CAMPANA, I.COURTOIS, C.FEDELICH and B.SANDOZ for their collaboration. Data for asymptomatic subjects were obtained thanks to ANR support (ANR_06_0385 SECUR ENFANT project).

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The Relationship Between Hip Flexion/Extension and the Sagittal Curves of the Spine C DRISCOLL1,2, C-E AUBIN1,2, H LABELLE2, J DANSEREAU1 1 – Dept. of Mechanical Engineering, École Polytechnique de Montréal P.O. Box 6079, Station Centre-Ville Montréal, Québec, H3C 3A7, Canada 2 – Saint-Justine University Hospital Center 3175 Cote-Ste-Catherine Rd. Montreal, Quebec, H3T 1C5, Canada Abstract. The objective of this study was to develop a finite element model (FEM) in order to study the relationship between hip flexion/extension and the sagittal curves of the spine. A previously developed FEM of the spine, rib cage and pelvis personalized to the 3D reconstructed geometry of a patient using biplanar radiographs was adapted to include the lower limbs including muscles. Simulations were performed to determine: the relationship between hip flexion / extension and lumbar lordosis / thoracic kyphosis, the mechanism of transfer between hip flexion / extension and pelvic rotation, and the influence that knee bending, muscle stiffness, and muscle mass have on the degree to which sagittal spinal curves are modified due to lower limb positioning. Preliminary results showed that the model was able to accurately reproduce published results for the modulation of lumbar lordosis due to hip flexion; which proved to linearly decrease 68% at 90o of flexion. Additional simulations showed that the hamstrings and gluteal muscles were responsible for the transmission of hip flexion to pelvic rotation with the legs straight and flexed respectively, and the important influence of knee bending on lordosis modulation during lower limb positioning. The knowledge gained through this study is intended to be used to improve operative patient positioning. Keywords: surgical positioning, lower limbs, spine, scoliosis, biomechanics, finite element modeling.

1. Introduction Patient positioning is increasingly being recognized as an important step in spinal surgeries [1,2,3]. One of the most important desired outcomes of spinal instrumentation surgeries is preservation / restoration of sagittal balance [4], which can improve long term fusion results [5,6]. The relationship between hip flexion and lumbar lordosis has been studied by Stephen et al. [7] on the Andrews frame and Befanti et al. [8] on the Wilson frame who concluded that that hip flexion results in a loss of lumbar lordosis and that the correlation between the two is subject to inter-patient variability. As a general rule hips should not be flexed greater than 30o in order to maintain sufficient lumbar lordosis to avoid flat back symptoms. The influence of lower limb positioning on thoracic kyphosis has received little attention. The mechanism of transfer between

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lower limb positioning and resulting geometrical changes to the spine has not been properly defined. Stokes et al. [9] have reported on the influence of the hamstring muscles on lumbar lordosis in sitting while others have reported on the possible contribution of the gluteal muscles with knees flexed [10]. Finite element modeling (FEM) has been previously used to study the impact of patient positioning on the geometry of the vertebral column. Duke et al.2 looked at the impact of dynamic positioning parameters on the geometry of the vertebral column using a FEM, the impact of lower limb positioning was approximated through the modulation of pelvic inclination +/- 15o. Our overall objective is to develop a patient-specific FEM which is able to simulate the impact of lower limb positioning on the geometry of the spine. It is hypothesized that manipulation of lower limb position, hip and knee flexion/extension, can significantly impact lumbar lordosis and thoracic kyphosis. It is also hypothesized that the mechanism of transfer between lower limb positioning and pelvic angle modulation includes both the hamstring and gluteal muscles groups and is dependent on the degree of knee flexion. The specific objectives of this pilot study were to use the developed FEM to analyze the relationship between hip flexion/extension and the sagittal curves of the spine. 2. Materials and Methods A previously developed simplified global beam FEM of the spine, rib cage and pelvis [11] which uses a biplanar reconstruction technique [12] to obtain patient specific geometry was adapted to include the lower limbs. Complementary detailed geometry of the femur, tibia, and fibula was taken from Visible Human Project (VHP) data with scaling based on anthropological equations factoring height and sex13. A total of 31 muscles per leg were modeled with origins and insertions obtained by mapping of the coordinates defined by White et al. [14]. Lower limb muscle cross-sectional data and material properties were taken from literature [15,16]. Hip and knee joints were represented as well as ligaments. In all, the model contains 1790 nodes and 1247 elements (Figure 1).

Figure 1 - Frontal and Lateral Views of the FEM developed

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Simulations were performed in order to evaluate the model’s ability to reproduce published results for loss of lumbar lordosis due to hip flexion [5,6]. The geometric reconstruction of a healthy young male subject taken from the databases at SainteJustine University Hospital measuring 150 cm with an initial L1-S1 lordosis of 44 o and a T4-T12 kyphosis of 31o was used for this and subsequent simulations. Hamstring muscle initial strains were uniformly adjusted such that the percent loss of lumbar lordosis obtained through FEM simulations matched the average results obtained by Befanti et al [6] for 30o of hip flexion. These conditions were maintained and additional simulations performed for 60o and 90o hip flexion with the results compared to the averages obtained by Stephen et al.5 in similar conditions. A sensitivity study was performed in order to determine the relative impact that hamstring muscle initial strain (representing flexibility), cross-sectional area, and elastic modulus have on the degree to which hip flexion impacts the sagittal profile of the spine. 30o hip flexion simulations were performed while varying hamstring muscle initial strain 0-10%, doubling and halving cross-sectional area baseline values, and elastic modulus +/- 50% their baseline value. In order to evaluate the muscles responsible for transmission of lower limb positioning to pelvic rotation and the impact of knee flexion on this relationship, hip flexion / extension simulations were performed, over their respective ranges of motion [17], at intervals of 20o while maintaining the knees different degrees of flexion 0o, 30o, 60o, and 90o. In each case, the resultant impacts on lordosis, kyphosis, and lower limb muscle strains were recorded. Muscles with strains exceeding their initial strain were considered stretched and to contribute passively to the transfer of lower limb positioning to pelvic rotation. 3. Results The results of the model validation based on published references are outlined in Table 1. They were obtained for a uniform hamstring initial strain of 4%.

Hip Flexion 30° 60° 90°

L1-S1 Lumbar Lordosis Simulated Results Published values5,6 33° 33° 24° 23° 14° 15°

Table 1 - Validation of the relationship between hip flexion and loss of lumbar lordosis

The sensitivity study demonstrated that the most influential factor impacting the degree to which hip flexion impacts the sagittal profile of the spine is muscle flexibility; a 5% increase in hamstring initial strain caused an additional 9% reduction of lordosis at 30o of hip flexion. The impacts of muscle cross-sectional area and elastic modulus were less important; doubling cross-sectional area and elastic modulus respectively caused an additional 4% and 6% loss of lumbar lordosis. The muscles found to be responsible for the transmission of straight leg hip flexion to pelvic rotation were the hamstrings (semimembranosus, biceps femoris long

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head). Once knee flexion reached approximately 60o, the gluteals (gluteus medius) started increasingly contributing. The primary muscles responsible for the transmission of hip extension to pelvic rotation are the anterior thigh muscles (rectus femoris, satorius, vastus medialis, tensor fasciae latae) up until the maximum hip range of motion. The degree of knee flexion (0o to 90o) for 30o hip flexion simulations contributed to additional modification of lordosis of 14%. The impact of lower limb positioning (90o of flexion to 30o of extension, while maintaining the knees at 30o of flexion) on lordosis (L) and kyphosis (K) is outlined in Figure 2. 4. Discussion One aspect of the model, which remains to be validated, is its ability to accurately reproduce the impact of hip extension on spinal geometry. Since no literary data was available, anterior thigh muscles were given the same initial strain determined for thehamstring muscles. Due to the nature of beam FEMs not all origin and insertion coordinates coincided with an anatomical representation, specifically with regards to the un-represented ilium, and had to be translated to the nearest available element. However, this factor was assumed to negligibly impact the results as the translation distances did not exceed 4 cm. Also it is important to note that only passive contribution of the lower limb muscles was considered as once a patient is place on a surgical frame, the current position can be maintained and variations to the lower limb position can be made without inducing voluntary or involuntary muscular contractions. In addition with any spinal surgical procedures, the patient is under general anaesthesia.

L = 48 K = 29 L = 45 K = 30

L = 42 K = 31

L = 33 K = 32 L = 23 22 K = 33 L = 13 12 K = 35

Figure 2 - Impact of Lower Limb Positioning

The results obtained for the mechanism of transfer between hip flexion and pelvic rotation is in agreement with those proposed in literature. This study was able to show which muscles of the hamstring and gluteal groups are responsible for this transmission and at what degree of knee flexion does the transmission from the hamstrings to gluteals take place. The inter-patient variability found in literature with regards to lumbar lordosis modulation with hip flexion can be attributed to individual lower limb flexibilities. It is proposed that any attempt made to predict patient specific modulation of spinal

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geometry due to lower limb positioning incorporate clinical flexibility testing data into personalized FEMs. 5. Conclusion The FEM developed has shown good agreement with published results in its ability to reproduce the impact of lower limb positioning on spinal geometry in a patient-specific manner. Its preliminary exploitation has allowed for a more detailed study of the mechanisms of transfer and influential factors between lower limb positioning and spinal geometry than has been previously reported in literature. It is believed that the use of surgical frames which allow lower limb positioning in conjunction with knowledge of how lower limb positioning impacts patient specific spinal geometry can be used in order to facilitate an improve upon current operating procedures. Acknowledgements Project funded by the Natural Sciences and Engineering Research Council of Canada (Industrial Research Chair Program, with Medtronic). References [1] [2] [3]

[4] [5]

[6] [7] [8]

[9] [10] [11] [12]

[13] [14] [15]

Schonauer C, Bocchetti A, Barbagallo G, Albanese V, Moraci A. Positioning on surgical table. Eur Spine J. 2004 Oct;13 Suppl 1:S50-5. Epub 2004 Jun 22. Review. Duke K, Aubin CE, Dansereau J, Labelle H. Computer simulation for the optimization of patient positioning in spinal deformity instrumentation surgery. Med Biol Eng Comput. 2008 Jan;46(1):33-41. Delorme S, Labelle H, Poitras B, Rivard CH, Coillard C, Dansereau J. Pre-, intra-, and postoperative three-dimensional evaluation of adolescent idiopathic scoliosis. J Spinal Disord. 2000 Apr;13(2):93101. Majdouline Y, Aubin CE, Robitaille M, Sarwark JF, Labelle H. Scoliosis correction objectives in adolescent idiopathic scoliosis. J Pediatr Orthop. 2007 Oct-Nov;27(7):775-81. Edwards CC, Levine AM. Complications associated with posterior instrumentation in the treatment of thoracic and lumbar injuries. In: Garfin SR, ed. Complications of Spine Surgery. Baltimore: Williams and Wilkins, 1989:164-99. Herkowitz HN. Lumbar spinal stenosis: Indications for arthrodesis and spinal instrumentation. Instructional Course Lectures 1994:43:425-33. Stephens GC, Yoo JU, Wilbur G. Comparison of lumbar sagittal alignment produced by different operative positions. Spine. 1996 Aug 1;21(15):1802-6; discussion 1807. Benfanti PL, Geissele AE. The effect of intraoperative hip position on maintenance of lumbar lordosis: a radiographic study of anesthetized patients and unanesthetized volunteers on the Wilson frame. Spine. 1997 Oct 1;22(19):2299-303. Stokes IA, Abery JM. Influence of the hamstring muscles on lumbar spine curvature in sitting. Spine. 1980 Nov-Dec;5(6):525-8. Tafazzoli F, Lamontagne M. Mechanical behaviour of hamstring muscles in low-back pain patients and control subjects. Clin Biomech (Bristol, Avon). 1996 Jan;11(1):16-24. Clin J, Aubin CE, Labelle H., Virtual prototyping of a brace design for the correction of scoliotic deformities. Med Biol Eng Comput. 2007 May;45(5):467-73. Delorme S, Petit Y, de Guise JA, Labelle H, Aubin CE, Dansereau J. Assessment of the 3-D reconstruction and high-resolution geometrical modeling of the human skeletal trunk from 2-D radiographic images. IEEE Trans Biomed Eng. 2003 Aug;50(8):989-98. Bhasin MK, Malik SL, editors. Anthropology: Trends and Applications. New Delhi: Kamala-Raj Enterprises, 2002. p.141-147. White SC, Yack HJ, Winter DA. A three-dimensional musculoskeletal model for gait analysis. Anatomical variability estimates. J Biomech. 1989;22(8-9):885-93. Klein Horsman MD, Koopman HF, van der Helm FC, Prosé LP, Veeger HE. Morphological muscle and joint parameters for musculoskeletal modelling of the lower extremity. Clin Biomech (Bristol, Avon). 2007 Feb;22(2):239-47. Epub 2006 Nov 28.

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[16] Kovanen V, Suominen H, Heikkinen E. Collagen of slow twitch and fast twitch muscle fibres in different types of rat skeletal muscle. Eur J Appl Physiol Occup Physiol. 1984;52(2):235-42. [17] Luttgens, Kathryn and Hamilton, Nancy. Kinesiology : Scientific Basis of Human Motion. Madison, WI : Brown & Benchmark, 1997

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Development of an Apparatus to Evaluate Adolescent Idiopathic Scoliosis by Dynamic Surface Topography T.M.L. Shannon Department of Computer Science, School of Technology, Oxford Brookes University. Oxford, U.K. Abstract. For cases of Adolescent Idiopathic Scoliosis (AIS), commonly the first indicator is a change in the surface shape of the back over time. A proportion of patients so diagnosed require surgical intervention to prevent further progression and to improve cosmesis. The results of a preliminary literature survey have revealed that significant work has already been published on the static acquisition and analysis of back surface shape. There is new interest in establishing correlations between breathing, posture, the underlying spinal deformity and changes in the surface topography of the back during clinical sessions together with an increased focus on the impact of the cosmetic defect on the patient and in the measurement of pre and postoperative dynamic capability. The continuing development of an apparatus based on established optical motion capture technologies, that generates a sequence of tri-dimensional images and provides measurements derived from changes in the position of anatomical reference landmarks and of the surface topography of the back will be presented. If, using the same landmarks, the trunk range of motion could be captured concurrently, it is hoped that the resulting data would form the basis of a useful clinical study.

1.

Introduction

Between 1984 and 1988, the author was involved with the development and introduction of the commercial version of the Integrated Shape Imaging System (I.S.I.S.) [1] based on the previous work of Turner-Smith and Houghton [2, 3]. For the past twenty years he has concentrated on the development and application of optical motion capture technologies to the fields of clinical gait analysis, rehabilitation, sports biomechanics and ergonomics. Within the last decade, the same technology has been widely applied to the creation of animated background characters used in the crowd scenes of many major film productions including Titanic, Troy, Gladiator, Star Wars II and III. In recent years the technology has advanced to a degree where it is now being used to capture the subtleties of characteristic whole body motion and facial expressions of well known actors as the main characters in feature films such as The Polar Express and Beowulf. The aim of the study was to investigate how it might be possible to apply similar technologies and experience to synchronously capture video images to quantify, in three dimensions, the changes in the position of surface anatomical landmarks and back surface shape during clinical presentations.

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Earlier work by many researchers concentrated on attempting to reduce the radiographic exposure to AIS patients by investigating if there was a reliable correlation between the progression of an underlying spinal deformity and changes in back surface topology over time [4,5,6]. The techniques applied and the relational algorithms developed were found to be prone to error by postural and non-spinal artifacts and were in-sufficiently robust to accommodate all cases so limiting their measurement sensitivity, specificity and usefulness as general clinical tools. A recent paper [7] focussed on the need to quantify the cosmetic defect of spinal deformity and work has been published on the evaluation of tri-planar spine range of motion following spinal fusion [8], implying a need still exists for apparatus [9] that can dynamically acquire the location of pre-defined anatomical surface landmarks and back topology to within clinically acceptable accuracies.

2. Materials and Methods Previous work using optical motion capture technology has been published. Rotelli and Santambrogio [10] placed an array of passive detectable markers across the surface of the back and captured the resulting tri-dimensional positions. Aliverti et al. [11] used a laser scanning mechanism to apply a moving point of light to the surface synchronized to each acquisition of the apparatus optical sensors. Rotelli and Santambrogio’s method had the advantage of presenting an absolute measure of the location of all markers during each acquisition but would not be a feasible option for routine clinical sessions due to the time needed to apply sufficient markers before each measurement. The approach by Aliverti et al. would have been prone to errors introduced by postural and breathing artifacts, so was not considered further. 2.1. Data Acquisition The study used an obsolete and modified 6 Camera, VICON motion capture system (Vicon Motion Systems Ltd., Oxford, U.K.) to acquire anatomical landmark positions and surface data simultaneously at a rate of 60 frames/second. Fig. 1 depicts the arrangement of two groups of three, optically isolated, cameras and a projector used to generate an array of points. VICON systems use spherical markers coated (Fig. 2) with a material that reflects light directly back to a strobed source surrounding a camera lens. The cameras are shuttered to open only during a strobe flash and contain filters optically matched to the spectra of the light source. Only circular bright markers will be sampled by each camera sensor, independent of the rate of subject movement and ignoring skin, fabric and other objects within the field of view. The centre of markers are calculated as positions within the two dimensional image illuminating the sensor during a given frame by analyzing the relative intensities of light impinging onto groups of adjacent pixels. Before each capture session, calibration objects with markers attached at known positions are used to establish the global coordinate system of the measurement volume; the physical position and orientation of each camera and the scaled

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relationship between the acquired coordinates and the actual positions. The optical distortion of the cameras (particularly from the lenses at the corners) is also calculated and a linearization correction applied to each subsequent frame captured. Within the measurement volume defined by the lenses chosen and the distance to the object, accuracy of marker centre reconstruction was determined by experiment to be within a mean of ± 0.1mm, S.D. 0.3mm, n = 4,500.

Fig. 1. Acquisition Apparatus

Fig. 2. Anatomical Markers

Fig. 3. Projected Point Cloud

To detect the markers, three shuttered, strobing cameras were configured to emit and detect light in the visible red spectral region (660 nm). Three further passive cameras were fitted with optical, short-pass filters to exclude light in the red spectra but to allow passage of the image of the projected surface point cloud (Fig. 3) to the camera sensors. The centres of the points were determined in the same way as for the markers. Providing a minimum of two calibrated cameras see a marker or a point anywhere within the measurement volume, the third dimension can be calculated in exactly the same way as humans estimate distance with two offset eyes viewing a common object [12]. The presence of a third or more cameras further improves the reliability of point and marker re-construction. 2.2. Surface Data Processing Figures 4 and 5 depict the presentation of anatomical marker (Fig. 4) and surface point cloud (Fig. 5) locations in two dimensions, acquired from a single frame from cameras in each group. Supporting software has been developed to automatically separate marker and point data into two files for further processing into frames of three– dimensional coordinates. The resulting three dimensional data were normalised to a reference plane defined by the positions in each frame of the markers placed by palpation over the vertebra prominens (C7/T1) and the left and right posterior superior iliac spines (PSIS) [1]. Two additional markers were applied to the acromion to provide a measure of shoulder droop. For pre-operative cases, 10-13 additional markers were applied to the spinous

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processes and a tri-planar cubic spline interpolation calculated to define the delineation between the left and right sagittal surface sections. Placement of markers in postoperative cases should follow the convention established by Jefferson et al. [13]. Boundary cubic splines were also calculated based on a proportion of the height of the spine between C7/T1 and the bisection point between the PSIS to remove data not required for further analysis such as the arms. Algorithms have been developed that automatically identify the reference markers and order spinous process markers in most expected cases within defined degrees of freedom. Table 1 lists the clinical data that are calculated and Fig. 6 depicts an example graph of changes in imbalance over 440 frames (7.3 s) captured from a normal subject.

Fig. 4. Anatomical Markers

Fig. 5. Surface Cloud Points

Prior to export for display and surface data interpolation, the resulting point cloud data were automatically allocated into one of twenty transverse sections above the normalised reference plane to further amplify volumetric differences between the sagittal sections. A number of third party surface topography interpolation and display packages (Surfer 8 © and Voxler ©, Golden Software Inc.) are currently being investigated for suitability. Fig. 7 depicts the resulting Voxler © output for frame 45 of a 150 frame capture of the subject’s back. Further work is being undertaken to use the results obtained to develop numerical measures to describe volumetric asymmetries. 2.3 Range of Motion Data Processing Range of motion data were captured from the same subject using common marker placements by switching off the point array projector and switching on the strobes (in the blue spectra) of the three surface capture cameras to improve the reconstruction accuracy of more rapidly moving markers within the measurement volume. Fig. 8 depicts the display of markers in three dimensional space and Fig. 9 a graphical sample of the angle subtended between C7/T1, the central spinous process marker and the marker adjacent to the PSIS over 2000 frames (33 s). The impact of skin movement of the markers on results will be considered as part of the final study.

T.M.L. Shannon / Development of an Apparatus to Evaluate Adolescent Idiopathic Scoliosis

Parameter Height Tilt Imbalance Pelvic Tilt Pelvic Obliquity Shoulder Droop

Units mm degrees (+/-) mm (L/R) degrees (L/R) degrees (to L/R) mm (L/R)

Table 1. Calculated Parameters

Fig. 6. Variations in Imbalance from a Normal Subject over 7.3 s.

Fig. 6. Variations in Imbalance from a Normal Subject over 7.3 s

Fig 7. Voxler© Data and Contour Image - Frame 45

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Fig. 8. Markers Single Fame

Fig. 9. Range of Motion Angles

3. Conclusion Development of the apparatus and supporting algorithms continues with the goal of producing a tool to quantify and express changes in back surface shape and the range of motion during a clinical session. The resulting data are hoped to form the basis of a useful clinical study.

References [1]

Turner-Smith A.R., Shannon T., Houghton G.R., Knopp D. Assessing idiopathic scoliosis using a surface measurement technique. Surgical Rounds for Othopaedics, 1988, June:5263. [2] Turner-Smith A. A television/computer three-dimensional surface shape measurement system. Journal of Biomechanics,1988,21(6):515-529 [3] Tuner-Smith A.R., Harris J., Houghton G.R., Jefferson R.J. A method for analysis of back shape in scoliosis. Journal of Biomechanics,1988,21(6):497-509 [4] Weisz I., Jefferson R.J., Turner-Smith A.R., Houghton G.R., Harris J. ISIS scanning: a useful assessment technique in the management of scoliosis. Spine, 1988:13(4):405-408. [5] Turner-Smith A.R. Surface topography should not replace radiography in the evaluation of scoliotic deformities. Proceedings of the International Symposium on 3-D Scoliotic Deformities, Dansereau J. 1992,Gustav Fischer Verlag: 178-180. [6] Stokes A.I.F. Point of View: Early Detection of Progression in Adolescent Idiopathic Scoliosis by Measurement of Changes in Back Shape with the Integrated Shape Imaging System. Spine, 1997:22(11):P1228. [7] Theologis T.N., Jefferson R.J., Simpson A.,Turner-Smith A.R. ,Fairbank J.C.T., Cosmesis in Adolescent Idiopathic Scoliosis. Proceedings of the 10th Philp Zorab Symposium, 2002 [8] Engsberg J., Kenke L., Reitenbach A., Hollander K. et al., Prospective evaluation of trunk range of motion in adolescents with idiopathic scoliosis undergoing surgery. Spine 27(12):1346-1354. [9] De Wilde T.,Huysmans T.,Denis K. Forausberger C. et al. A contact-free optical measuring system for the dynamic acquisition of anatomical structures in 3D.Proceedings of the 14th Conference of the European Society of Biomechanics, 4-7 July 2004. [10] Rotelli F., Santambrogio G.C., Three-Dimensional Dynamic Reconstruction of Back Shape. Three Dimensional Analysis of Spinal Deformities, M.D. D’Amico et als.(Eds), IOS Press, 1995:45-50.

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[11] Aliverti A., Ferrigno G., Rotelli F., Santambrogio G.C., Back Surface Analysis by Laser Beam Scanning and Stereo-Photogrammetry. Three Dimensional Analysis of Spinal Deformities, M.D. D’Amico et als.(Eds), IOS Press, 1995:51-56. [12] HartleyR.I.,Zisserman,A.,Multiple View Geometry in Computer Vision, 2nd Edition, 2004, Cambridge University Press, ISBN: 0521540518. [13] Jefferson R.J., Weisz I., Turner-Smith A.R., Harris J.D., Houghton G.R. Scoliosis Surgery and it’s Effect on Back Shape. J Bone Joint Surg [Br],1988;70-B:261-6.

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Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved. doi:10.3233/978-1-58603-888-5-133

133

SpineCor vs. Natural History – Explanation of the results obtained using a simple biomechanical model H-R WEISS Asklepios Katharina Schroth Spinal Deformities Rehabilitation Centre, Korczakstr. 2, 55566 Bad Sobernheim, Germany, [email protected] Abstract. In the recent peer reviewed literature the SpineCor is described as an effective method of treatment for patients with scoliosis. However until recently no prospective controlled end-result study is presented comparing the results obtained with this soft brace to natural history. The objective was to determine whether the results obtained by the use of the SpineCor are better than natural history during pubertal growth spurt. The method employed prospective comparison of the survival rates of SpineCor treatment vs. natural history with respect to curve progression during pubertal growth spurt. 12 Patients with Cobb angles between 16 and 32 degrees (at average 21 degrees) during pubertal growth spurt are presented as a case series treated with the SpineCor. Survival rate of this sample is described and compared to natural history (SRS brace study 1995). All girls treated in both studies were at risk for being progressive with the first clinical signs of maturation (Tanner 2-3). During the pubertal growth spurt most of the patients (11/12) with SpineCor progressed clinically and radiologically as well (at least 5 degrees). Progression could be stopped changing SpineCor to the Chêneau brace in most of the sample described (7/10). The avarage Cobb angle at the start of treatment with the SpineCor was 21.3 degrees, after an average observation time of 21.5 months 31 degrees. At 24 months of treatment time 33% of the patients with the SpineCor where still under treatment with their original bracing concept, at 72 months follow-up time 8 % of the patients with the SpineCor survived with respect to curvature progression. Survival proportion in the SpineCor sample, though was 0.08, while in the natural history cohort it was 0.34. The SpineCor treatment during pubertal growth spurt seems to lead to a worse outcome than observation only. The use of a simple biomechanical model explains that in the brace the compression forces exceed the lateral forces used for the corrective movement. Therefore SpineCor does not seem to be indicated as a treatment during pubertal growth spurt. Keywords. Adolescent idiopathic scoliosis, SpineCor, natural history, growth spurt

1.

Introduction

In the recent peer reviewed literature the SpineCor is described as an effective method of treatment for patients with scoliosis [1,2]. However until recently no prospective controlled end-result study is presented comparing the results obtained with this soft brace to natural history. Two studies reveal data that support the hypothesis, that the use of the SpineCor brace is less successful than the use of other braces during pubertal growth spurt [3,4].

134

H.-R. Weiss / SpineCor vs. Natural History

Figure 1. SpineCor as adjusted by the original authors [1,2] at our centre. No changes have been made to the original adjustments unless a clear progression has been detected.

Objectives: If the use of the SpineCor during pubertal growth spurt is less successful than the use of other braces, the question remains to be answered as to whether one can expect at least results superior to natural history using this soft brace. To determine whether the results obtained by the use of the SpineCor are better than natural history during pubertal growth spurt, this study has been undertaken.

2.

Material and Methods

This is a prospective comparison of the survival rates of SpineCor treatment vs. natural history with respect to curve progression during pubertal growth spurt. 12 Patients with Cobb angles between 16 and 32 degrees during pubertal growth spurt are presented as a case series treated with the SpineCor. Survival rate of this sample is described and compared to natural history as presented in the control group of the SRS brace study [5]. All girls treated in both studies were at risk for being progressive with the first clinical signs of maturation (Tanner 2-3), the girls from the SpineCor sample all were premenarchial at the start of treatment.

3.

Results

During the pubertal growth spurt most of the patients (11/12) with SpineCor progressed clinically and radiologically as well (at least 5 degrees). Progression could be stopped changing SpineCor to the Chêneau brace in most of the sample described (7/10). The avarage Cobb angle at the start of treatment with the SpineCor was 21.3 degrees, after an average observation time of 21.5 months 31 degrees. At 24 months of treatment time 33% of the patients with the SpineCor where still under treatment with their original bracing concept, at 72 months follow-up time 8 % of the patients with the SpineCor survived with respect to curvature progression. Survival proportion in the SpineCor-sample, though was 0.08, while in the Natural History cohort it was 0.34.

H.-R. Weiss / SpineCor vs. Natural History

135

SRS study finished

Observation (estimated)

SpineCor final result

6,0 Years

Figure 2. Survival analysis comparing natural history [modified from 5] to SpineCor treatment (fat grey line) during pubertal growth spurt clearly shows worse results for the SpineCor sample at 4 years.

4.

Discussion

In this study no patients have been lost for follow-up. Therefore the results of our study can be compared to other studies with similar study design [4]. The material in the study performed by Nachemson et al. [5] however, is slightly different including also postmenarchial girls (65 premenarchial, 46 postmenarchial) with clear signs of maturation (82 with Risser 0-1, 28 with Risser 2-5). Follow-up time is not shorter in our study compared to Nachemsons (72 vs. 48 Month) and the last remaining patient (18 years of age) in our study was no more of risk for progression at the end of the observation period. The proportion of patients without progression (rate of success) under Boston brace treatment was 74 % (worst case 50%) whereas the Chêneau sample from our previous study did quite better with a success rate of 80% [3] (no patient lost, one patient Risser 1, the others Risser 0 in the Chêneau sample of patients). Success rate for the SpineCor sample was 8% compared to the success rate of the natural history sample in the Nachemson study [5] of 34%. So we cannot expect the SpineCor treatment to change natural history, although we must admit the SpineCor sample to have a slightly worse prognosis (being more immature) than the controls [5]. The poor results of SpineCor treatment during pubertal growth spurt may be explained by the fact, that this soft brace leads to axial loading (Fig. 3.) with kyphosing effects to the thoracolumbar junction as shown in lateral x-rays [2], while sagittal realignment with lumbar re-lordosation has been shown to be beneficial in the treatment of patients with scoliosis [6-8] and therefore this should be implemented in actual bracing concepts.

5.

Conclusions

The SpineCor treatment during pubertal growth spurt seems to lead to a worse outcome than observation only. The use of a simple biomechanical model explains that in the

136

H.-R. Weiss / SpineCor vs. Natural History

Figure 3: In a sphere wrapped into elastic material the compression forces can be estimated to be the same at every point (left). In a lengthwise oval model (right) – independently of any lateral forces applied – axial compression forces will always overweight the lateral forces leading to a junctional thoracolumbar kyphosis with the consequence of an instable segmental alignment in this area.

brace the compression forces exceed the lateral forces used for the corrective movement. Therefore SpineCor does not seem to be indicated as a treatment during pubertal growth spurt.

References [1] [2] [3] [4]

[5]

[6] [7] [8]

Coillard Ch, Leroux MA, Zabjek KF, Rivard Ch: SpineCor – a non-rigid brace for the treatment of idiopathic scoliosis: post-treatment results. Eur Spine J 12:141-148, 2003. Coillard Ch, Circo AB, Rivard Ch: SpineCor treatment for early scoliosis: 15 to 25°. 5th. Internationel Conference on the Conservative Management of Spinal Deformities, Athens, April 2-5, 2008 Weiss HR, Weiss GM: Brace treatment during pubertal growth spurt in girls with idiopathic scoliosis (IS): a prospective trial comparing two different concepts. Pediatr Rehabil. 2005 Jul-Sep;8(3): 199-206. Wong MS, Cheng CY, Ng BWK, Lam TP, Sin SW, Lee-Shum LF, Chow HK, Tam YP: A prospective study on clinical efficacy and patients´ acceptance of SpineCor and rigid spinal orthoses in treatment of AIS. 5th. Internationel Conference on the Conservative Management of Spinal Deformities, Athens, April 2-5, 2008 Nachemson AL, Peterson LE: Brace study group of SCR. Effectiveness of treatment with a brace in girls who have AIS. A prospective, controlled study based on data from the brace study of the SRC. J Bone Jt Surg, 77-A:815-822, 1995. Weiss HR, Dallmayer R, Gallo D: Sagittal counter forces (SCF) in the treatment of idiopathic scoliosis: a preliminary report.Pediatr Rehabil. 2006 Jan-Mar;9(1):24-30. Weiss HR, Rigo M: The chêneau concept of bracing - actual standards.Stud Health Technol Inform. 2008;135:291-302. van Loon PJ, Kühbauch BA, Thunnissen FB: Forced lordosis on the thoracolumbar junction can correct coronal plane deformity in adolescents with double major curve pattern idiopathic scoliosis. Spine, 2008 Apr 1;33(7):797-801.

Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved. doi:10.3233/978-1-58603-888-5-137

137

Comparison of the kyphosis angle evaluated by video rasterstereography (VRS) with x-ray measurements H-R WEISS, N ELOBEIDI Asklepios Katharina Schroth Spinal Deformities Rehabilitation Centre, Korczakstr. 2, 55566 Bad Sobernheim, Germany, [email protected] Abstract. Surface topography evaluations are prone to technical errors due to postural sway of the patients measured. The technical error of lateral deviation (rms) and surface rotation (rms) may vary between 15 and 20%, while the kyphosis angle (IP-ITL) has a technical error of only 5% (2,5 degrees), which is comparable to the x-ray measurement. Purpose of this study was to investigate the hypothesis that video rasterstereography can be used for prognostication of a kyphosis patient. Materials and Methods. 53 Patients (23 females, 30 males, average age 17 years with a range from 11 to 56 years) undergoing in-patient rehabilitation have been measured with the help of video rasterstereography (VRS) before starting the treatment program and the values for kyphosis angle have been correlated to the kyphosis angle measured on a lateral x-ray (XR) not older than 6 weeks before VRS measurement. 26 had a thoracic Scheuermann, 3 a thoracolumbar, 15 an Idiopathic Kyphosis and 9 a kyphosis of other origin. Results. Average Kyphosis angle XR was 49 degrees (SD 17) and VRS 63 degrees (SD 13). There was a high significant Pearson correlation of 0.78 and a high significant difference of 14 degrees in the t-test (t –9,6, p<0,001). Conclusions. The kyphosis angle VRS (Vertebra prominens – lower neutral zone of inclination) seems to allow a follow-up of individual kyphosis patients. The XR kyphosis angle according to Stagnara is measured from T4 to the lower end vertebra and therefore is lower than the VRS kyphosis angle measured from T1. The difference found between XR and VRS kyphosis angles may be explained by the angle between T1 (VRS) and T4 (XR) differently used as the upper end vertebra. Therefore the prognostication of an individual patient seems possible within certain limits. Keywords. Kyphosis angle, videorasterstereography, VRS, radiography

1.

Background

Surface topography evaluations are prone to technical errors due to postural sway of the patients measured. The technical error of lateral deviation (rms) and surface rotation (rms) may vary between 15 and 20% [1,2], while the kyphosis angle (IP-ITL) has a technical error of only 5% (2,5 degrees), which is comparable to the x-ray measurement [3]. Purpose of this study was to investigate the hypothesis that video rasterstereography can be used for prognostication of a kyphosis patient. 2.

Material and Methods

53 Patients (23 females, 30 males, average age 17 years with a range from 11 to 56 years) undergoing in-patient rehabilitation have been measured with the help of video

138

H.-R. Weiss and N. Elobeidi / Comparison of the Kyphosis Angle Evaluated by VRS

rasterstereography (VRS) before starting the treatment program and the values for kyphosis angle have been correlated to the kyphosis angle measured on a lateral x-ray (XR) not older than 6 weeks before VRS measurement. 26 had a thoracic Scheuermann, 3 a thoracolumbar, 15 an Idiopathic Kyphosis and 9 a kyphosis of other origin.

Th 4

Th 1

Figure 1. The XR kyphosis angle according to Stagnara is measured from T4 to the lower end vertebra and therefore is lower than the VRS kyphosis angle measured from T1.

3.

Results

Average kyphosis angle XR was 49 degrees (SD 17) and VRS 63 degrees (SD 13). There was a high significant Pearson correlation of 0.78 and a high significant difference of 14 degrees in the t-test (t –9,6, p<0,001). 4.

Discussion

The kyphosis angle VRS (Vertebra prominens – lower neutral zone of inclination) seems to allow a follow-up of individual kyphosis patients. The XR kyphosis angle according to Stagnara is measured from T4 to the lower end vertebra and therefore is lower than the VRS kyphosis angle measured from T1. The difference of at average 14 degrees found between XR and VRS kyphosis angles may be explained by the angle between T1 (VRS) and T4 (XR) differently used as the upper end vertebra. Therefore the prognostication of an individual patient seems possible within certain limits using VRS. VRS kyphosis angles therefore seem to be precise and can be used for prognostication and follow-up of kyphosis patients as well.

H.-R. Weiss and N. Elobeidi / Comparison of the Kyphosis Angle Evaluated by VRS

139

15 degrees should be subtracted from the VRS (VP – ITL) kyphosis measurements to estimate the XR Stagnara angle of kyphosis. 5.

Conclusions

The kyphosis angle VRS (Vertebra prominens – lower neutral zone of inclination) seems to allow a follow-up of individual kyphosis patients. The XR kyphosis angle according to Stagnara is measured from T4 to the lower end vertebra and therefore is lower than the VRS kyphosis angle measured from T1. The difference found between XR and VRS kyphosis angles may be explained by the angle between T1 (VRS) and T4 (XR) differently used as the upper end vertebra. Therefore the prognostication of an individual patient seems possible within certain limits. References [1]

[2]

[3]

Weiss, HR, K. Lohschmidt, N. El Obeidi: The Automated Surface Measurement of the Trunk. Technical Error. In: J.A. Sevastik and K.M. Diab (Eds.): Research into Spinal Deformities I, IOS Press, 1997, pp: 305-308 Weiss, HR, K. Lohschmidt, N. El Obeidi: Trunk Deformity in Relation to Breathing. A Comparative Analysis with the Formetric System. In: J.A. Sevastik and K.M. Diab (Eds.): Research into Spinal Deformities I, IOS Press, 1997, pp: 323 – 326 Weiss, H.R., Dieckmann J, Gerner, J.: The practical use of surface topography: following up patients with Scheuermann’s disease. Pediatr Rehabil. 2003 Jan-Mar;6(1):39-45

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Postural changes in patients with scoliosis in different postural positions revealed by surface topography K SCHUMANN, I PÜSCHEL, A MAIER-HENNES, H-R WEISS Asklepios Katharina Schroth Spinal Deformities Rehabilitation Centre, Korczakstr. 2, 55566 Bad Sobernheim, Germany, [email protected] Abstract. Claims have been made that surface topography is an objective tool, however there are significant postural influences (relatively large technical error due to postural sway) those measurements are prone to. Purpose of this study was to help estimate these influences by measuring patients with scoliosis in three standardized postural positions. Material and Methods. We studied the surface-topography measurement in 100 inpatients with idiopathic scoliosis divided into different age-groups. First group: 7 to 12 years (n=12), second group: 13 to 16 years (n=51), the third 17 to 20 years (n=15) and the fourth > 21 years (n=22) (7 males and 93 females). The thoracic Cobb angle was 26.4 degrees, lumbar Cobb angle 25.7 degrees. We investigated the average lateral deviation (rms) and average surface rotation (rms). Measurements were taken one day before the patients left the clinic, after a 3 or 4 week in-patient intensive rehabilitation program (SIR), in three different postures: Normal posture: no specific instructions: standing with feet in an standardized way. Conscious posture: The patients acquired this posture during intensive daily exercising. Corrected posture: The most corrected posture the patients are able to achieve by using specific muscle tension and specific breathing techniques. We compared the results between the different postures. Then we calculated the results for the different age groups. Results. There are significant differences in both parameters tested, some of them more than 40% to 67% greater than the measurement error calculated. The best results were achieved in the second and the third group with the conscious posture, the adult group had the best valued in most corrected posture. For the youngest patients there were no significant changes with the different postures. Conclusions. Surface measurements can be influenced by artificial postures and therefore cannot be attributed as objective. This is why the surface measurements should be made by someone independent from the treatment process in order to exclude any bias as far as possible. Surface topography may be used for postural monitoring in the rehabilitation process of patients with scoliosis. Keywords. Videorasterstereography, VRS, rehabilitation, postural control, objectivity

1.

Background

Claims have been made that surface topography is an objective measurement, however there are significant postural influences (relatively large technical error) that influence such measurements [1-3]. The purpose of this study was to help estimate these influences by measuring patients with scoliosis in three standardized postural positions.

K. Schumann et al. / Postural Changes in Patients with Scoliosis in Different Postural Positions

2.

141

Material and Methods

We studied the surface topography measurement in 100 in-patients with idiopathic scoliosis divided into different age-groups. First group: 7 to 12 years (n=12), second group: 13 to 16 years (n=51), the third 17 to 20 years (n=15) and the fourth > 21 years (n=22) (7 males and 93 females). The thoracic Cobb angle was 26.4 degrees, lumbar Cobb angle 25.7 degrees. We investigated the average lateral deviation (rms) and average surface rotation (rms). Measurements were taken one day before the patients left the clinic, after a 3 or 4 week in-patient intensive rehabilitation program (SIR), in three different postures: Normal posture: No specific instructions: standing with feet in an standardized way. Conscious posture: The patients acquired this posture during intensive daily exercising. Corrected posture: The most corrected posture the patients are able to achieve by using specific muscle tension and specific breathing techniques. We compared the results between the different postures. Then we calculated the results for the different age groups.

3.

Results

There are significant differences in both parameters tested, some of them more than 40% to 67% greater than the measurement error calculated. The best corrective effects were achieved in the second and the third group with the conscious posture. The adult group had the best values in most corrected posture. For the youngest patients there were no significant changes with the different postures. The results are listed in table 1 and 2. Group

age

NP

ADLP

CP

I II III IV

10,8 (SD 1,7) 14,6 (SD 1,0) 17,7 (SD 1,0) 39,8 (SD 12,9)

11,1 (SD 6,7) 11,4 (SD 6,0) 13,8 (SD 5,3) 17,2 (SD 6,8)

10,1 (SD 6,6) 8,9 (SD 4,8) 11,1 (SD 4,8) 14,9 (SD 7,5)

10,6 (SD 5,6) 8,3 (SD 4,4) 11,2 (SD 4,7) 12,8 (SD 6,3)

Table 1. Average lateral deviation (rms) in the four different age groups in “normal” (scoliotic) posture (NP), ADL posture (ADLP) and in corrected posture (CP).

Group

age

NP

ADLP

CP

I II III IV

10,8 (SD 1,7) 14,6 (SD 1,0) 17,7 (SD 1,0) 39,8 (SD 12,9)

5,6 (SD 2,1) 6,8 (SD 3,0) 7,4 (SD 2,8) 8,7 (SD 2,9)

6,3 (SD 4,1) 6,2 (SD 2,8) 6,5 (SD 2,3) 7,7 (SD 2,8)

6,2 (SD 1,9) 7,3 (SD 4,5) 7,9 (SD 3,6) 9,0 (SD 3,2)

Table 2. Surface rotation (rms) in the four different age groups in “normal” posture (NP), ADL posture (ADLP) and in corrected posture (CP). The changes achieved here are not as clear as the changes in lateral deviation.

142

4.

K. Schumann et al. / Postural Changes in Patients with Scoliosis in Different Postural Positions

Discussion

With the outcomes for the different age groups, we are able to create a more evidence based model for conservative treatment in order to improve postures that enable patients to stop the vicious cycle of scoliosis progression by a more balanced loading (unloading) of spine.

5.

Conclusions

Surface measurements can be influenced by artificial postures and therefore cannot be attributed as objective. This is why the surface measurements should be made by someone independent from the treatment process in order to exclude any bias as far as possible. Surface topography may be used for postural monitoring in the rehabilitation process of patients with scoliosis [4-7]. The limitations of the use of surface topography to document the outcome of patients during conservative management have well been described [8].

Figure 1. Surface reconstruction of a patient in her scoliotic posture (NP) on the left and corrected during an exercise to achieve best corrected posture (CP) in daily activities.

References [1] [2]

[3]

[4]

Weiss, H.R., Dieckmann J, Gerner, J.: The practical use of surface topography: following up patients with Scheuermann’s disease. Pediatr Rehabil. 2003 Jan-Mar;6(1):39-45 Weiss, HR, K. Lohschmidt, N. El Obeidi: The Automated Surface Measurement of the Trunk. Technical Error. In: J.A. Sevastik and K.M. Diab (Eds.): Research into Spinal Deformities I, IOS Press, 1997, pp: 305-308 Weiss, HR, K. Lohschmidt, N. El Obeidi: Trunk Deformity in Relation to Breathing. A Comparative Analysis with the Formetric System. In: J.A. Sevastik and K.M. Diab (Eds.): Research into Spinal Deformities I, IOS Press, 1997, pp: 323 - 326 Weiss, HR, Ch. Verres, N. El Obeidi: Ermittlung der Ergebnisqualität der Rehabilitation von Patienten mit Wirbelsäulendeformitäten durch objektive Analyse der Rückenform. Phys Rehab Kur Med 9 (1999) 41-47

K. Schumann et al. / Postural Changes in Patients with Scoliosis in Different Postural Positions [5] [6]

[7]

[8]

143

Weiss HR, Steiner A, Reichel D, Petermann F, Warschburger P, Freidel K. Medizinischer Outcome nach stationärer Intensivrehabilitation bei Skoliose. Phys Med Rehab Kuror 2001;11:100-103 Weiss, HR, Ch. Verres, K. Steffan, I. Heckel: Outcome Measurement of Scoliosis Rehabilitation by Use of Surface Topography. In: I.A.F. Stokes (Hrsg) Research into Spinal Deformities 2, IOS Press 1999, pp 246-249 Rigo M, Quera-Salvá G, Villagrasa M, Ferrer M, and Casas A: Effect of specific exercises on the sagittal profile of scoliotic spines. 4th International Conference on Conservative Management of Spinal Deformities, Boston, MA, USA. 13–16 May 2007. Scoliosis 2007, 2(Suppl 1):S7 Weiss HR: Conservative treatment effects in spinal deformities revealed by surface topography – a critical review of literature. 5th. Internationel Conference on the Conservative Management of Spinal Deformities, Athens, April 2-5, 2008

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Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved. doi:10.3233/978-1-58603-888-5-161

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A Novel Solution for Registration of Stereo Digital Torso Images of Scoliosis Patients 1

A KUMAR1, N DURDLE1 J RASO2 Dept of Electrical & Computer Eng, University of Alberta, Edmonton, AB, Canada 2 Glenrose Rehabilitation Hospital, Edmonton, AB, Canada Abstract. This paper presents a procedure for registration of a pair of stereo digital images giving an improvement in accuracy and speed over existing methods. It does so by a novel approach combining color based image segmentation and differential geometry. It involves three stages: image segmentation, adaptive local pixel matching, and deferential geometry in a tree weighted belief propagation procedure. The registration was compared to 2 existing registration procedures, segment-based adaptive belief propagation (adaptive BP) and color-weighted hierarchical belief propagation (hierarchical BP). A 3D scan of a mannequin was obtained and errors in reconstruction were measured for each of the 360 cross sections of the mannequin. The proposed procedure outperforms existing methods, particularly for high curvature regions and significantly large cross sections. Its accuracy of reconstruction ranged from 85-100% compared to 75-100% for other existing methods. It was 35% to 40% faster. This work provides a solution to the registration problem and is an important step in developing a cost effective technique for measuring torso shape and symmetry of scoliosis patients using stereo digital cameras. Keywords. Scoliosis, Registration, Image Segmentation, Pixel Matching, Belief Propagation

1.

Introduction

Scoliosis is a condition characterized by lateral deviation of the spine coupled with rotation of individual vertebra resulting in visible torso asymmetries [10]. Assessment of the severity of scoliosis is traditionally done using radiographs of the spine [11]. However, radiographs do not describe the visible torso deformity associated with scoliosis. Torso imaging can describe torso deformities and reduce the need for radiographs, effectively reducing the risk of cancer [11]. Optical/Imaging methods have been used for assessment of scoliosis and monitoring of its progression. Some of these are Moiré topography, ISIS scanning, Quantec system scanning, rasterstereography, laser scanning and stereo vision imaging [11]. Much advancement has been made in improving each of these methods over the past years. However, ISIS scanning is a slow process and produces poor resolution images. The Quantec system scanning, laser scanning and rasterstereography are expensive due to the hardware components involved and also immobile. Images produced via Moiré topography are highly dependent on posture of the patient and provide a relative measure based on contours rather than a quantitative measure. In light of the problems associated with 3D imaging of the torso, stereo vision has become a cost effective alternative. Progress in computer vision and availability of faster computer processors has lead to development of stereo vision algorithms for simultaneous stereo camera capture, calibration and reconstruction. Stereo capture systems consist of digital or TV cameras

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A. Kumar et al. / A Novel Solution for Registration of Stereo Digital Torso Images

positioned with known geometry. Significant advances have been recently made in the area of computer vision, as a result of publically available performance testing such as the Middlebury data set [1], which has allowed researchers to compare their algorithms against all state-of-the-art algorithms [7]. Stereo correspondence or registration of stereo images is one of the most active research areas in computer vision [1]. In the area of stereo vision research, stereo images refer to images captured at different viewpoints using cameras with known geometry [5]. In order to register the stereo images, we need to determine the closest (least error) or best point-to-point correspondence between the two images. Stereo registration algorithms can be used on stereo images captured using calibrated digital or TV cameras to obtain three dimensional (3D) point set data. A triangulation algorithm is primarily applied on point set data to obtain a 3D surface. 3D surface reconstructions of scenes or localized objects in a scene using stereo vision has modern applications in 3D modelling, computer graphics, facial expression recognition, surgical planning, architectural structural design etc. [2,8]. The 3D scene geometry established from this process can be used to reconstruct 3D torso images to study scoliosis. However a problem associated with the existing stereo registration algorithms such as that developed by Klauss, Sormann and Karner [4] is that it assumes frontal parallel plane geometry. This means that it assumes depth is constant (with respect to the rectified stereo pair) over a region under consideration [3]. Reconstruction of smooth and curved surfaces where depth is constantly changing violates this assumption. Li and Zucker [8] have tried to solve this problem using differential geometry but they have not applied their algorithm to the one developed by Klauss, Sormann and Karner [4]. Belief Propagation and Graph Cuts are the most commonly used methods for refinement of the registration process [1]. Tree Re-weighted Message Passing, which might become a serious rival to Belief Propagation and Graph Cuts [13], has not been used with differential geometry. Tree Reweighted Message Passing’s improvement over Belief Propagation and Graph Cuts becomes significant for more difficult functions [13]. This paper presents a procedure for registration of a pair of stereo digital images using image segmentation, adaptive local pixel matching, and differential geometry in a tree reweighted belief propagation procedure. The major contribution of this paper lies in incorporating differential geometry and Tree Reweighted Message Passing to Segment-Based Stereo Matching Using Belief Propagation and a Self-Adapting Dissimilarity Measure [4] to create a novel procedure with better accuracy and speed. 2.

Materials and Methods

Stereo registration methods are assessed using univalued disparity function of one image with respect to the other (referred to as reference image). When first introduced in human vision literature, disparity was used to describe the difference in location of corresponding features seen by the left and right eye [1]. The x, y spatial coordinates of the disparity space are taken to be coincident with the pixel coordinates of a reference image. The correspondence between a pixel in a reference image and pixel in the matching image is linearly related to each other by the disparity function. The disparity function obtained over the 2D images is known as the disparity space image (DSI). DSI gives a 3D point set data using the calibration

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parameters of the stereo cameras. It represents the confidence or log likelihood of a match implied by the disparity function. The procedure for obtaining disparity is divided into three stages: mean shift color segmentation, adaptive local pixel matching, and differential geometry in a tree reweighted belief propagation procedure as illustrated in Figure 1.

STEREO REGISTRATION PROCEDURE

Mean Shift Color Segmentation

Adaptive Local Pixel Matching

Differential Tree Reweighted Belief Propagation

Figure 1. Flow-chart of stereo registration procedure.

The process of color segmentation is to decompose the reference image into regions of color or grayscale [4]. The mean-shift analysis approach is essentially defined as a gradient ascent search for maxima in a density function defined over a high dimensional feature space [4]. Comaniciu and Meer’s [13] mean shift segmentation is insensitive to differences in camera gain and is therefore used. The next step involving local pixel matching is an essential step for defining a disparity plane. The aim of this step is to provide an initial estimate of the disparity space image (DSI). This basic idea is to obtain a measure of disparity that can be optimized and refined. The disparity plane is based on 3D x-y-d space supporting slanted and curved surfaces where x, y are spatial coordinates and d is the inverse depth or disparity. The disparity planes are calculated using local pixel matching. Local pixel matching requires calculation of a matching score and an aggregation window [4]. Matching score is obtained using a self-adapting dissimilarity measure that combines the sum of absolute pixel intensity differences (SAD) and a gradient-based measure as implemented by Klauss, Sormann and Karner [4]. The last stage involves calculation of the final disparities. The algorithms that perform well in this stage are based on an energy minimization framework. This means that we need to choose at each pixel the disparity associated with the minimum cost value [1]. It involves minimizing two separate energy functions that are summed together to calculate the final energy minimization term as given by the equation, E(d) = Edata(d) + λEsmooth(d) (1) where d represents disparity. The symbol Edata(d) measures how well the disparity function agrees with the input image pair and is given by the summation of matching score over the spatial coordinates. The formulation of Edata(d) is Edata(d) = Σ C(x, y, d(x, y)) (2) where C is the matching score. The symbol Esmooth(d) encodes smoothness in the image by measuring the differences between the neighboring pixels’ disparities [1]. It is given by the following equation,

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Esmooth(d) = Σ ρ(d(x, y) − d(x+1, y)) + ρ(d(x, y) − d(x, y+1)) (3) where ρ is some monotonically increasing function of disparity difference. The Esmooth(d) operates on the frontal parallel assumption that is altered in this procedure as suggested by Li and Zucker using “floating” disparities [2]. Li and Zucker apply “Floating disparities” on the Max-Product Belief Propagation framework. TreeReweighted Message Passing as defined by Kolmogorov [13] is used in this procedure as the energy minimization framework. The key subroutine of the Tree-Reweighted Message Passing algorithm is Max-Product Belief Propagation [13]. The “floating” disparities [2] are therefore added to the Max-Product Belief Propagation component of Tree-Reweighted Belief Propagation. The Tree-Reweighted Belief Propagation is advantageous because messages are passed in a sequential order rather than a parallel order requiring half the space. Convergence is reached in two passes rather than having a convergence condition as in the case of Max-Product Belief Propagation. Therefore applying differential geometry to Tree-Reweighted Belief Propagation produces better results. Stereo digital images of 400x300 resolution from the Middlebury dataset [1] and the resultant DSI obtained using the above procedure are shown in Figure 2 below.

a

b

c

Figure 2. A. Digital Image from left camera. B. Digital Image from right camera. C. Disparity Space Image taking image from left camera as reference image and image from right camera as matching camera.

3.

Results

The registration was compared to 2 existing registration procedures, segment-based adaptive belief propagation (adaptive BP) and color-weighted hierarchical belief propagation (hierarchical BP). A 3D scan of a mannequin was obtained and errors in reconstruction were measured for each of the 360 cross sections of the mannequin. The proposed procedure outperforms existing methods, particularly for high curvature regions and significantly large cross sections. Its accuracy of reconstruction ranged from 85-100% compared to 75-100% for other existing methods. It was 35% to 40% faster. 4.

Discussion

This work provides an improvement over existing stereo correspondence methods. It is an important step in developing a cost effective technique for measuring torso shape

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165

and symmetry of scoliosis patients. Future work will focus on validating the procedure on larger and more complex datasets. References [1] [2] [3] [4] [5] [6] [7]

[8] [9]

[10] [11]

[12] [13]

Scharstein D Szeliski R A Taxonomy and Evaluation of Dense Two-Frame Stereo Algorithms Int J Computer Vision, 2002 47(1/2/3):7-42. Li G Zucker SW “Surface Geometric Constraints for Stereo in Belief Propagation. Computer Vision and Pattern Recognition, 2006 IEEE Computer Society Conference 2006 2 2355 - 2362 Li G Zucker SW. Differential Geometric Consistency Extends Stereo to Curved Surfaces, In: Proc. 9-th European Conference on Computer Vision ECCV 2006 (3) 44-57 Klaus A Sormann M Karner K Segment-Based Stereo Matching Using Belief Propagation and a SelfAdapting Dissimilarity Measure, Pattern Recognition, ICPR 2006, 3,: 15-18 Sun J Zheng N-N Shum H-Y Stereo Matching Using Belief Propagation. Pattern Analysis and Machine Intelligence, IEEE Transactions, 2003 25, (7) 787 - 800 Bleyer M Gelautz M Graph-based surface reconstruction from stereo pairs using image segmentation Proceedings of the SPIE, 2004 5665, 288-299 Yang Q Wang, L Yang R Stewenius H Nister D Stereo Matching with Color-Weighted Correlation, Hierarchial Belief Propogation and Occlusion Handling, Computer Vision and Pattern Recognition, IEEE Computer Society Conference 2006 2, 2347- 2354 Li G Zucker SW. Stereo for Slanted Surfaces: First Order Disparities and Normal Consistency Proc. EMMCVPR, LNCS, 2005 - Springer Zhang L Curless B Seitz SM. Rapid Shape Acquisition Using Structured Light and Multi-pass Dynamic Programming First International Symposium on 3D Data Processing Visualization and Transmission (3DPVT'02) 2002. 24 Ajemba PO Durdle NG; Hill DL Raso VJ. Re-positioning Effects of a Full Torso Imaging System for the Assessment of Scoliosis. Canadian Conf on Electrical and Computer Eng. 2004, 3, 1483-1486 Ajemba PO Durdle NG; Hill DL Raso VJ. A Torso Imaging System for Quantifying the Deformity Associated with Scoliosis Instrumentation and Measurement, IEEE Transactions. 2007 56, Issue 5, Oct. 1520 - 1526 Kolmogorov V. Convergent Tree-Reweighted Message Passing for Energy Minimization, IEEE Transactions on Pattern Analysis and Machine Intelligence, 2006 28, 10 Comaniciu D; Meer P. Mean Shift: A Robust Approach Toward Feature Space Analysis, IEEE Transactions on Pattern Analysis and Machine Intelligence, 2002 24 (5) 603-619

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Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved. doi:10.3233/978-1-58603-888-5-170

The central cord-nervous roots complex and the formation and deformation of the spine; the scientific work on systematic body growth by Milan Roth of Brno (1926-2006) P. J.M. van Loon1 and L W. van Rhijn2 1

Slingeland Hospital, Doetinchem, The Netherlands Department of Orthopaedic Surgery, University Hospital Maastricht, The Netherlands

2

1. Introduction and biography. Professor Milan Roth died on April 4, 2006. He was a professor of neuro-radiology of the J.Ev.Purkynẽ-University (now: Masarek-University) in Brno, Czechoslovakia, now the Czech Republic. Milan Roth was born on October 6, 1923 in Lelekovice, a small village north of Brno, in local schoolmaster's family. After completing his grammar school studies in Brno in 1942, he spent a year improving his language skills because the Czech universities were closed at the wartime. Since 1943 till the end of World War II, he was a "Totaleinsatz" in an armament factory in Kurim near Brno. After the war, he studied at the Medical Faculty of (now)Masaryk University in Brno, completing his studies on September 29,1949. After completing his studies, he was assigned to the department of surgery at the local hospital in Brunntal in northern Moravia (till January 31, 1950). During 1950-1952 he served in the army, most of the time at the Department of Radiology of the Military Hospital in Plzen (Pilsen). After completing his military service, he spent a short time at the pulmonary department of the local hospital in Prerov, and then at the Central Department of Radiology of the Medical Faculty in Olomouc (now Palacky University- both Prerov and Olomouc are located in central Moravia, northeast of Brno). Since November 1, 1954, he has been with the Central Department of Radiology of the Medical Faculty in Brno (now Masaryk University) that became Department of Radiology and Nuclear Medicine in 1960, and this was his workplace till his retirement in August1989. He had an appointment in research too, in the field of anatomy. In this part of his working live he produced his impressive work, both quantitative and without doubt now also qualitative. After his retirement on the University he stayed a part-time radiologist of the Radiodiagnostic Clinic of the hospital in Brno-Bohunice. He suffered a brain stroke early in 1999, became seriously afatic. Just before the planned visit of the first author,

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his medical condition detoriated quickly. Milan Roth passed away peacefully amidst his family on April 4 2006. His work remained largely unnoticed in the mainstream of the international world of orthopaedics, neurology or neurosurgery.

Fig .1 Milan Roth at the age of 80.

Roth had a major disadvantage in exporting his knowledge to a more global scientific forum, that a large part of his career, while developing and unfolding his ideas and concepts, he worked behind the Iron Curtain. Only in his early career he worked as a fellow, not accompanied by his young family as that was forbidden in the communistic regime, at the Karolinska Institute of the University in Stockholm. His analytical work was first mainly published in German and Czech language, and in publications not well noticed because inaccessible for the mainstream. Only in the beginning of the 1980`s he had a few articles in English more international orientated journals in the field of Radiology. . At the end of this contribution a bibliography of selected articles is added with the main purpose to disclose them to a wider audience. His main concept is the concept of skeletal morphogenesis he developed which is based on the existence of 2 different types of growth of the spinal column, viz., the cellular-divisional (mitotic) growth of bone and soft tissues and the extensive (stretch) growth of the nervous structures. The neural extensive growth proceeds at a slower rate than the bone growth. Which is manifested most conspicuously in the ascent of the spinal cord. In his concept a number of normal as well as pathological features of the skeletal morphogenesis- above all the gross deformities of the body-parts which grow rapidly in length such as the spine, the extremities or the facial skeleton – are explained by the disturbed relation of the two growth types caused by an insufficiency of the vulnerable neural extensive growth. 2. Overview of the work of Roth We are not able to specify, discuss or review all his work in this contribution. It can however be divided it in four main chapters:

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2.

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4.

Analytical work, undertaken mainly during the decade 1960-1970, in which he laid the fundamental of the research examining and integrating interpretation of older work in the fields of biology, embryology, anatomy and physiology. Developing mechanical models of the spine, spinal growth and deformations in order to explain and visualise the processes of interrelated types of growth in the growing body in regard of time and the processing of forces. Proposing the integrated concept of neuro-osseous growth and its role in explaining the normal development of tissues creating the organs and organic systems, viewing the spine as a crucial part of the skeleton Published articles in the 1970 and 1980`s in which he consequently built upon his integrated concepts. This included papers on topics such as the Arnold Chiari malformation and Syringomyelia, and also the role and development of the neural foramen and the deformations in the lumbosacral spine based on secondary changes, like stenosis. These subjects are not discussed in the present paper.

This paper will consider this contribution focussing mainly on the developing of Roth’s integrated concept of neuro-osseous growth which was published in several papers the early nineteen-seventies. 2.1. Analytical work Roth’s knowledge of the superb work of European scientists on the spine, which he often quoted, was prodigious, even with his insight into their disputing certain concepts. An increasing number of great scientists were investigating medical problems in Europe in the early twentieth century, which included spinal deformations, with names such as Lorenz, Nicolodani, Albert and Schulthess noted. They conducted anatomical studies, mainly on cadaver vertebrae and discs. Roth was the first to correlate their individual findings from their painstaking anatomical study with his own discoveries resulting from the early application of pneumencephalo- and myelography to the position, calibre, direction and suspected tension of the central cord and vertebral roots in normal and scoliotic spines. He stated: “The traction effect of the spinal nerves is of decisive influence upon the position of the spinal cord within the spinal canal as well as upon the shaping of the vertebral foraminae, a conclusion we reached in our own radiographic studies, published in 1965 and 1966. 2.2 Phylogenetic work. The hypothesis that the tensionless position of the central nervous system matures into the adult animal vertebrate body was demonstrated in worms and other, more higher developed animals as well as in radiological and cadaver human studies. By applying study to the normal state allowed an development of understanding of the requirements of the central nervous tissues d to grow and develop. 2.3 Experimental teratogenic work. In order to understand the role biochemistry plays (and still plays) in the development of a range of deformities of the spine, extremities and jaw, Roth conducted experimental studies on teratogenesis to clarify the role of the up to then neglected osteo-neural growth patterns, an area in which the interaction of growth in the nervous

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system interacts with other tissues. . Roth was able to impair the stretching properties of the nervous system by exposing it ( intra-uterinal) to either teratogenic substances or by lowering the level of oxygen within it. In these experiments, he was able to create spinal deformities as scoliosis. His experiments demonstrated that the so-called passive role of neurogenic tissue, especially in the central cord, was transformed into a major, active role during the development and growth of the spine and the extremities. This ability of nervous tissue to grow by extension is phylogenetically determinate. Growth and errors of growth of the vertebrate spinal skeleton was thus removed, allowing the “intimate relation existing from the very beginning of development between the nervous and the bony tissue to continue working incessantly during the whole growth period of life”. 2.4 Mechanical models of the spine, spinal growth and deformations. With his interests in the growth and deformation of the spine and knowledgeable in related fields of biological or medical science, Roth developed his own mechanical models for different issues in a quest for an better understanding of the development of the normal, as well as the distorted or deformed spine. He was fully aware that forces in nature are often invisible, such as the growth and tensions within the body. He realised that only the consequences of these forces on a body can be seen and thus a conceptual clarification of the universally adopted relationship between form and function (ie acting forces) is essential in order to understand these important processes in nature. In an paper on the models of vertebro-neural relations, Roth described an elastic model which could dynamically reproduce the physiological relationships of the growth of osseous structures with that of nervous tissue. Using Plexiglas, rubber, textile and metal he was able to construct an accurate model representing all the components of his concept of proportioned and disproportional growth between different tissues. The dynamic role played by normal living, the tensions created by growth, all superimposed on the pressures present in all living structures maintaining an upright position or on the pretension existing before making any movements, were developed in a range of experiments. Roth created all his models himself. He fully realised that there were contrasts between the early embryological patterns and the later adult state. Radiographs were made of these models in different stages of applied forces of tension, using different planes in order to demonstrate the close similarities with the radiographic images of living individuals. The model also explained the ascent of the conus medullaris during growth, widely seen then as a passive displacement. In some pathological conditions, like myelomeningocele, the ascent is said to be hindered by a tight string of fibrous tissue, the filum terminale, causing a “tethered cord”. Roth claimed that the less elongated or more stretched and tense spinal cord and associated attached roots would explain the impossibility of it “ascending” properly into the position of the conus at the T12-L1 area whereas in other animals the conus is low in the lumbar spine. The ascent was thus seen as an adaptation to the prevailing upright position of the body in mankind. The so-called “tethering” is thus not a passive binding by strings, but an intrinsic quality of nervous tissue. The stressed lordotic component of the thoracolumbar spine required in order to create optimal biomechanical and neuromechanical conditions is also a very interesting aspect of his models.

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Fig 2 A photograph of a single segment of the thoracic spine in the transversal plane with modelling of the ribs, roots and cord. From: Roth M. [Models of vertebro neural relations]. Cesk Radiol 1970 Sep; 24(5): 189-94. (Courtesy of Roth’s heirs).

Fig 2b The whole construct of spine, inhabitants and ribs in two different positions distinguished by a certain amount of tension in the combined system of different tissues. From: Roth M. [Models of vertebro neural relations]. Cesk Radiol 1970 Sep; 24(5): 189-94. (Courtesy of Roth’s heirs).

For his concept of the osteo-neural growth concept, Roth devised a very simple model, made from beads on a string. This “necklace” model, which can be easily copied today with materials such as slightly deformable beads, gives an insight in what can happen when nervous tissue is unable to keep up to the extending energy growing bone is delivering. The tension within the bones or the counteracting energy of

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muscle actions occurring as a result of activity act as supercharged engine dictated by nervous tissue itself. Normal growth leads to the development of slender and long bones, but as the trend towards lengthening of bones takes place, the resulting retardation of extension of nervous tissue leads to the beads becoming compressed and thereby deformed. 2.5. The concept of osteo-neural growth. The underlying reason for the focus of Roth’s scientific work on the relationship between the central nervous system and the osseous components of, the spine, is not made clear. The quest must have been strong enough to allow him unrestrained research across all fields of biological and medical science. Certainly the existence of scoliotic curves in the human spine, not encountered in other animals, puzzled him as much as it still does today, concentrating his mind in his attempts to clarify the neglected role of the central spinal cord. Furthermore, the biomechanical adage that form always follows or reflects function was a mainstay in his thinking throughout his published work. What is certain is that attention was drawn to the relationship between nervous tissues and the spinal canal by his pioneering work as a neuroradiologist, employing the techniques of pneumencephalography, myelography and positive contrast examinations of the scoliotic spine. 2.6 Biological studies The comparison of the growth of animal nervous tissue by extension and the growth of central fibres in plants has already been fully explained and is undeniable. It has also been demonstrated that there is no cell proliferation of nervous tissue after birth in animals. Nervous tissue, in vertebrates as in all other animals, must possess an alternate pattern of growth, unlike all other tissues in the body. These grow or renew themselves at all stages of life, by rapid and intensive cell proliferation during growth. ``As in trees, the growth pattern is by extension of the cells (extensive growth; German: Streckungswachstum). Roth compared the governing phytohormon auxine, a trypthophane derivate driving this type of growth in plants which is said to be highly susceptible to a number of growth inhibitors (Gutenberg, Lehrbuch der Botanik) with the role of serotonine in the growth of nerves in animals. The energy necessary to stretch cells is generated by tensile power in the surrounding tissues during growth. In the spine, the vertebrae and discoligamentary complex, growing by cell proliferation, is responsible for the energy needed to stretch associated nervous tissues: “tag on tow”. With his interest in biology and, specifically the biological literature relating to plant and animal growth, Roth adapted the law of cephalocaudal differential growth in which growth in animals, just as in plants, is found to be directed from the first embryological components to develop such as the central tissue of an animal. In the vertebrate, the central cord, in the early embryological stages, is advanced when compared to the first segmentation of adjacent tissues. Although describing a leading role for the osseous structures to play by acting in the lengthening of the central nervous structures, by application of the law of cephalocaudal differential growth, Roth placed the phylogenetically oldest part of nervous system, the central cord and brainstem, as an initiating, architectural and controlling component and contributor to the lay-out of the surrounding tissues. These in turn form the basis of the biomechanical properties of the even more intriguing spine.

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Fig 3. A schematic drawing of the necklace model in which prolonged or progressive tension on the string gives deformation of the vertebrate skeleton like beads. From: Roth M.: The relative osteo-Neural growth; a concept of Normal and pathological (Teratogenic) skeletal Morphogenesis.Gegenbauer’s morph. Jahrb. Leipzig 119 (1973)2, S.250-274 (courtesy of Roth’s heirs)

Fig. 4 A schematic drawing showing the difference in how tissues in vertebrate animals grow, namely by cell proliferation comparing the epiphyseal zones around a schematic joint at the left, and the elongation by stretching of a none proliferating nervous tissue cell. (From: Roth M. [Idiopathic scoliosis--a special type of osteo-neural growth disproportion]. Z Orthop Ihre Grenzgeb 1969 Nov;107(1): 37-46). Courtesy of Roth’s heirs.

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3. The leading role of the nervous tissue in morphogenesis of the axial skeleton. Roth stresses that, prior to his work (and not really changed subsequently), the primary role and place of the nervous tissue in development and growth of the body in the context of other systems, such as the vascular, gastrointestinal or musculo-skeletal, was ignored. By demonstration of an exaggerated ventral-concave curvature (hyperkyphosis) of the early embryo, Roth referd to and used schematic drawings, with examples, of earlier work of Blechschmidt.

Fig. 5. Schematic drawing of embryological stages in which the enormous relative volume of the nervous system and the comma like shape is depicted, gradually changing to a stretched and more slender configuration, before the biomechanical needed cervical and lumbar lordosis is created. From: Roth M.: The relative osteo-Neural growth; a concept of Normal and pathological (Teratogenic) skeletal Morphogenesis.Gegenbauer’s morph.Jahrb., Leipzig 119 (1973)2, S.250-274 (courtesy of Roth’s heirs)

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When the ascent of the conus-cauda occurs, until now seen as a passive event, the associated lengthening of the spinal nerve roots are widely considered as being dragged along by the rapid growth of the spinal column. Roth, however, hypothesized an opposite view, suggesting that there is also a controlling role for the central nervous system in achieving the final form of the spine in adulthood. He compares it to the leading role the unrolling and growing brain substances play in the formation of the skull (calva) at the end of growth. Roth also explained why the conus in man is present at the thoracolumbar joint other than in the lower lumbar spine as it is in almost all other quadrupeds. 4. Summary It is clear from his writings that Roth applied his on-going practical experience of the clinical picture of children with deformations of the spine to his postulations. He observed that a normal lordotic shape of the lumbar and thoracolumbar spine is of great importance in allowing the correct functioning of the complexities of this area, the most mobile part of the spine in childhood. He described the requirement that the lower thoracic ribs should elevate, but also to retro-pulse, aided by the bellows-like function of the diaphragm. The tension placed on the neuromuscular structures can be measured by applying the mechanical law’s of Robert Hooke on properties of tensile bodies. However, while springs may be of value in understanding the complexities involved in biomechanics and nervous system control, this was not foreseen by Roth . The deformation of intervertebral discs and the osseous elements of the spine the result of the same forces which allow standing, sitting, balancing and movement. delivered by the muscular system. No muscle acts without nervous instruction. 5. The consequences of Roth’s work on scoliosis and practice. Roth applied his concepts and explanations of the mechanism of the deformities of: scoliosis. Roth stated that: “There exists a most intimate interrelationship between the rapid cranio-caudal growth-in-length of the axial skeleton and the slower, proceeding, extensive growth of the spinal cord and the nerve roots. The latter type of growth requires a greater energy supply and, consequently, is more vulnerable than the former one. The vertebro-neural growth relationship is similar to that existing between the developing brain and the skull. The growth in length of individual vertebrae, and of the spine as a whole, is adapted to the growth-potentiality of the intraspinal nervous structures, viz.; the former is determined by the availability of space among the latter. Idiopathic scoliosis may be interpreted as an adaptive morphogenetic reaction of the vertebral column to the growth insufficiency of the intraspinal nervous structures: The growth processes of the vertebral column, though continuing undisturbed at the cellular level, is adapted at the organ level by “waves” of the growth-insufficient cord-nerve complex. Morphological features of the scoliotic vertebra, coupled to the typical position of the spinal cord within the spinal canal, support the hypothesis of a vertebro-neural concept which offers a plausible explanation of both congenital and experimental scoliosis.” It might be argued that restoring the form e.g. by bracing; is not that harmful because it lengthens the spine, and thereby increases tension on the cord. Roth again provided an answer:

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“Adjustment or rectification of a deformed structure like a scoliotic spine is not accompanied by lengthening, notwithstanding the fact that the scoliotic trunk is elongated; the curved spine is adjusted, not lengthened. This adjustment involves a contraction or reduction of the convexe side of the intervertebral discs. Consequently in adjusting– paradoxally but irrefutably- the spinal canal is rather shortened.” [15]. 6. The work of Milan Roth: new challenges for future research Science is an ongoing process possessing its own dynamics. Several recent publications indicate that Roth’s hypotheses and concepts are now receiving attention. We are convinced that full disclosure and understanding of the work of Roth will inspire many of our colleagues to start new research and that these challenges will be taken up in many different fields of (medical) science. This will include: 1.) Reproducing and clarifying several of his analytical studies (pneumencapholgraphy) by using modern technologies such as MRI, Spiral CTscan. Also neuro-physiological studies, using SSEP and surface EMG, can be undertaken in situations with higher tensions in the neuromuscular structures. For instance, the recent publication of Cheng et al (2008) on the short spinal cord with scoliosis is evidence that this kind of work is already in progress. 2.) Research on what can cause differences in the quality of stretch growth of the cord in order to elicit if this is the background of the misunderstood variation of spinal forms such as curvature, disc-height, vertebral height and shape. Should genetic backgrounds also be investigated? 3.) A new coordinated effort between biomechanics and biochemistry to clarify the mechanism behind and between the growth (proportionate or disproportionate) of bone and tissues in relation with the stretch growth of the neural tissues. For biomechanics, following Roth, this includes the role of tension, tension patterns and tension regulation. For biochemistry, this requires research on the role of transmitters, such as serotonine, already implicated in a range of processes and diseases. What is their role, how do they work in the period of growth and is their function altered in body with deformations, even mild ones? How is the process of distribution of the lava-like material surrounding the notochord, as Roth calls it, organised? Roth concepts could give clues as to the final form of the body and organs. 4.) New concepts for treating patients by conservative or operative treatments with emphasis on the requirements for the nervous system as part of the complex “game” of compressive and tensile forces References Milan Roth: a selected list of his publications This bibliography with relevant publications by Roth consists of publications collected by the first author in recent years. Some of the articles were acquired from Roth’s family. The publications are presented in chronological order. To allow maximum disclosure all articles are presented in the following way: • An English version of the title • Statement of the language in which the original was published • An English summary or abstract (mostly original, sometimes extended) is added [1]

Roth, M (1965), Caudal end of the spinal cord, Acta Radiologica Diagnosis Vol. 3 (1965) Original Language: English

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P.J.M. van Loon and L.W. van Rhijn / The Central Cord-Nervous Roots Complex Abstract: The normal position of the spinal cord with special emphasis on the “dorsolumbar junction” as the part of the spine lacking a detailed knowledge of gross anatomic features as demonstrated at negative contrast myelography is discussed. Material consisted of 34 adults and 8 children without symptoms in this part or known remote disorders. The variation in position, the calibre and the intrinsic curvature of the cord at the different levels is explained by developmental factors, among which the morphology of the vertebrae is stressed. A cadaver study with fresh spinal cords specimen hanging freely showed a constant intrinsic curvature in the distal cord as a “reminder “of the embryological existent complete rounded configuration. Also the eccentric “lodgement” of the roots in the neuroforaminae is depicted as a consequence of the ascent of the cord, as seen in humans.

[2]

Roth, M. (1966), Vertebro-medullary interrelations as observed in gas myelography, Acta Radiologica Diagnosis Vol. 4 (1966), p. 569-580 Original language: English Abstract: The typical position of the spinal cord within the membranous sac, possibly derived from the close developmental relations between the neural tissue and the vertebral column, is described. The significance of the characteristic shape of the intervertebral foramina in predicting the depth of the ventral subarachnoid space is discussed. It is shown that small thoracic disk protrusions, with a narrow ventral subarachnoid space especially at the level of the lumbar intumescences, may give rise to myelopathy.

[3]

Roth, M. (1968), Idiopathic scoliosis caused by a short spinal cord, Acta Radiologica Diagnosis Vol. 7 (1968), p. 257-271. Original language: English Abstract: An explanation of the pathogenesis of idiopathic scoliosis based on the disturbance of the relative vertebro-neural growth is presented. This concept is supported by neuroradiologic, experimental and clinical observations.

[4]

Roth, M. (1969), Models of vertebro-neural relations, Acta Radiologica Diagnosis. Vol. 9 (1969) Original Language: English Abstract: The basic principles of vertebro-neural growth relations with special reference to the pathogenesis of idiopathic scoliosis are discussed. Plexiglas models, constructed to assist in the demonstration, are described. All models have in common that they do not only reflect a static threedimensional condition but represent the incorporated forces in life and growth by visible movements and represent also a condensation of what happens in the fourth dimension: time.

[5]

Roth, M. (1971), The relative osteo-neural growth-some phylogenetic, ontogenetic and clinical aspects, ad. Diagn. 1971, 1, p 81-97 Original language: German Abstract: The growth in length of the nervous structures necessitates a higher energetic level than that of the bones. A harmonic side-by-side growth course of the growth rates, cellular-divisional and neural-extensive, is indispensable for the normal body growth in length. Comparisons are made with features of growth in different animals and embryological knowledge. The relative osteo-neural growth represents therefore an important factor in the phylogenetic and ontogenetic development of the skeleton as well as in the pathogenesis of bone dysplasias.

[6]

Roth, M., The relative osteo-neural growth Part 1: Gegenbaur Morph. Jahrb. , Leipzig 117 (1971) 2, S. 232-255 Part II: Gegenbaur Morph. Jahrb. , Leipzig 117 (1972) 3, S. 312-334 Part III: Gegenbaur Morph. Jahrb. , Leipzig 117 (1972) 4, S. 421-440 Original language: German Abstract: These three articles compose a total concept and explanation of the osteo-neural growth by extracting supporting evidence out of first class previous research in biological, anatomical, embryological, histological and orthopaedic and neurological textbooks and literature. With own modelling, all sort of research, and stepping over of “scientific” boundaries, he tries to fit all visible formation and deformation of at least the skeletal development in his holistic explanation of how Nature “works”. In Part I the function of the Ascensus Medullae in Homo erectus is discussed.. The inhibititive (“braking”) power of the more energy asking stretch type of growth of the neural system on the growth by mitosis of the osseous and arthrogenic skeleton gives an insight in the complex system of tension regulation and strive for optimalisation growth in nature is. The earlier discovered and described craniocaudad and posteroanterior directed growth in ante- and postnatal growth in animals is completely incorporated in Roth’s conceptual thinking. The change in relative size of the primitive

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neural structures from huge to small is depicted and discussed as is the very clear present brachy-or Platyspondylie in siblings. The position, direction and calibre of the nerve roots in the neuroforaminae found at earlier pneumencephalographic studies at all different levels are made understandable. Different types of vertebral forms, especially the incisura vertebrae caudalis (roof of the foramen) are clarified. Different affections like Scheuermann, Platy- (or better: brachyspondylie, Dysplasia spondylo-epiphysealis tarda are according to Roth examples of clear “braking” of growth by hindered stretching capacity of the neural tissue. The formatted bone is like the narrow disc spaces diverged in horizontal directions. Roth destilles out of his daily practice, that the thoracolumbar junction is the most inflicted in flattening of the discs and vertebrae in adolescent kyphotic deformities. There fore he ends with the statement that the length of the total spine is fixed by the intraspinal nerve structures.

Fig.6 The formation of the pulmonary groove depends on the extension of the completely kyphotic and rolled up configuration of the embryonic body. Roth created a beautiful mechanical model on the change of all dimensions and relationships initiated by the gradual extension of the body.

Fig.7 The impressive relative change in proportions between cord and roots and vertebral bodies in animals, normal human spine, a scoliotic spine and a platyspondylic spine.

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P.J.M. van Loon and L.W. van Rhijn / The Central Cord-Nervous Roots Complex Part II. Roth tries to come to a complete system of growth and formation of the body especially the complete skeleton based on earlier work of scientists and own studies. What seems true for the vertebra seems true for every piece of bone.

Fig 8. Schematic drawing of different skeletal deformities with the suspected relation with the concomitant, but primary events in the stretching type of growth of the central cord. Part III. On the form of the body and the physiological curvatures of the spine out of animal experiments in rats and frogs: “The nervous structures exert an influence on the morphogenesis of the skeleton even for the very reason of their existence in space, by their mass of a certain size and shape. The surrounding and /or accompanying skeletal masses adapt their general shape to that of the nervous structures. obviously under mediation of a “trophic” neural effect. Through this primary neural influence the gross shape of the skeleton most appropriate for the given species in a given environment is elaborated. The stimuli and information reaching the organism by neural pathways are thus reflected in the morphogenesis of the skeleton .The role of the blood and the vessels (hormones-authors), of the physical stresses and of the hereditary (genetics- authors) appear only of secondary importance only in the light of osteo-neural growth relations”. [7]

Roth, M. (1973), The relative Osteo-Neural growth: a concept of normal and pathological (Teratogenic) Skeletal Morphogenesis, Gegenbauers morph. Jahrb. Leipzig 119 (1973) 2, S. 250-274 Original Language: German Abstract: A concept of skeletal morphogenesis is proposed which is based on the existence of 2 different types of growth in the vertebrate body, viz., the cellular-divisional bone growth and the extensive (stretch-) growth of the nervous structures. The latter type requires a higher energy and oxygen supply than the former. Consequently, the neural extensive growth proceeds at a slower rate than the bone growth. Which is manifested most conspicuously in the ascent of the spinal cord. The slower growth rate, however, is a general feature of the nervous structures throughout the vertebrate body. A number of normal as well as pathological features of the skeletal morphogenesis- above all the gross deformities of the body-parts which grow rapidly in length such as the spine, the extremities or the facial skeleton – can be readily explained by the disturbed relation of the two growth types caused by an insufficiency of the vulnerable neural extensive growth.

[8]

Roth, M. (1975), Spinal cord and Scoliosis. The cause and the Effect, Acta Chir. Orthop. Traumas. Czech. 42, 1975, no. 6, p. 507-517. Original language: Czech Abstract: There exist the most intimate interrelation between the rapid craniocaudad growth-in-length of the axial skeleton and the slower proceeding extensive growth of the spinal cord and the nerve roots. The latter type of growth requires a higher supply with energy and, consequently, is more vulnerable than the former one. The vertebro-neural growth relation is similar to that existing between the developing brain and its bony case. The growth in length of the individual vertebrae and of the spine as a whole is adapted to the growth-potentiality of the intraspinal nervous structures, viz.; the former is determined by the availability of space among the latter. Idiopathic scoliosis may be

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interpreted as an adaptive morphogenetic reaction of the vertebral column upon the growth insufficiency of the intraspinal nervous structures: The growth process of the vertebral column, though continuing undisturbed at the cellular level, is adapted at the organ level by “waves” to the growthinsufficient cord-nerve complex. Morphological features of the scoliotic vertebra together with the typical position of the spinal cord within the spinal canal speak in favour of the suggested vertebroneural concept, which offers a plausible explanation of the congenital and experimental scoliosis as well. [9]

Roth, M. , The vertebral groove, Acta Radiol.9; 1965 p. 740-745. Original language: English Abstract: Roth shows the presence, and gives name to the peculiar anatomical bilateral groove at the posterior surface of the bony spine. The development of the vertebral groove at the posterior side of the lamina as a unique feature in the human skeleton is explained and its influence via the spinal nerves on the shape of the intervertebral foramina is described. It fits in Roth’s view about the supposed working of the musculature to provide forces on bony structures that will add in their final shape as form is dictated by functional request. In the axiom that form follows function it is the far more posterior presence of the facet joints and processi mammilaris in human that is specific for the species and originates from the tremendous greater pulling forces of the musculus Erector Trunci in the osseous insertions in the upright man.

[10] Roth, M. (1969), Idiopathic scoliosis- A special form of osteo-neural growth disproportion Z Orthop Ihre Grenzgeb 1969 Nov;107(1):37-46) Original language: German Abstract: Idiopathic scoliosis is explained as a pathological increase of the vertebro-neural growth disproportion, the physiological degree of which is reflected in the ascent of the spinal cord. This disproportion roots in the two different types of growth occurring in vertebrates, viz., the cellulardivisional and the neural-extensive. The latter is generally encountered in plant-kingdom. The morphological findings on scoliotic vertebrae as well as model experiments point to the primary growth insufficiency of the intraspinal nervous structures as the actual cause of the idiopathic scoliosis. Roth, M., The experimental Teratogenesis of the skeleton. An experimental disturbance of the relative osteo-neural growth.Gegenbaur Morph. Jahrb. , Leipzig 122 (1976) 5, S. 686-730 Original language: German Abstract: The previously suggested concept of the closest growth relations existing between the bony and the nervous tissue at the organ level of the spinal cord and the peripheral (including the facial) nervous trunks is experimentally buttressed. It is shown that the normal gross-morphological features of the vertebrae as well as of the tubular bones (viz., their length, physiological curvatures and terminal expansions) result from the adaptation of the bone growth to the slower proceeding and vulnerable neural extensive growth, viz., from a physiological osteo-neural growth disproportion. The more or less conspicuous growth in the length of the facial skeleton depends upon the phylogenetically established, more or less evolved extensive-growth potentially of the facial nervous trunks as well. The growth relation existing between the developing brain and its bony case applies essentially even for the axial organ, the extremities as well as for the facial skeleton. The experimental findings speak in favour of the theoretical expectation that the typical teratogenic deformities of the extremities (micromelia), of the spine (scoliosis, defects of the vertebrae and of the ribs) as well as of the beak (jaws) which may be produced by a great number of most diverse teratogens, result from the adaptation of the bone growth to the growth-insufficient nervous trunks, viz., from the pathologically enhanced osteo-neural growth disproportion. The cleft palate and the digital defects (syndactylia, oligodactylia) may be readily explained by the growth-inhibition of the palate and digital nervous structures as well. The vertebrate body may be thus conceived as composed of 2 growth types, viz., the neural-extensive and the cellular-divisional (mitotic). The former is represented by an extremely dense felt work of nerve fibers and trunks (the Donaldson’s “ nervous skeleton”), which is “ stuffed “ with the other, mostly mitotic growing tissues. The 2 growth types are closely related partly at the macro- (organ-) level concerning the normal and teratogenic morphogenesis of the skeleton, partly at the micro-level of the utmost periphery, viz., of the terminal extensive meshwork and the individual cells or groups of cells. The cells which escape from the extensive felt work (i.e. from the “ nervous skeleton”) such as the superficial cells of the epidermis or mucous membranes and, in all probability, the elements of the haemopoetic organs, perish under normal conditions, suffer a planned, highly purposeful death. With regard to the lack of normal nerves within malignant tumors, the malignant cell may be conceived as

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[11] Roth, M., J. Krkoška and I. Toman Morphogenesis of the spinal canal, normal and stenotic, Neuroradiology 10, 277-286 (1976) Original language: English Abstract: The shape of the canal in transverse view, the shape and the position of the facet joints and the foraminate are discussed in normal and pathological conditions, like the developmental stenosis (Verbiest) and degenerative stenotic and degenerative changes are explained by the conductive role the neural tissues play in growth. Histological specimens are used. New models of the relationship between roots and vertebrae are introduced (fig). But nevertheless the base of the early base of any deformation seems still orchestrated by the necessary normal or disturbs the developmental balance between the two tissues (neural and osseous- discoligamentary). Roth states firmly in this paper that the role of the Notochord, a prominent structure in fishes, amphibians and reptiles is vestigial in higher mammals and its morphogenetic role in the developmental events of the axial skeleton is grossly overestimated (The Dispensability of the Notochord).

Fig 9 Modelling of the relationship of the roots, their directions and the presumed thight contact between the roots and the bony surroundings in the lumbar spine. [12] Roth, M. (1981), Idiopathic scoliosis from the point of view of the neuroradiologist, Neuroradiology (1981) 21: 133-138 Original language: English Abstract: There is a simple morphological interrelation between the growing spinal cord-nerve root complex and the vertebral column, not unlike that between the growing brain and the skull. The shape of the enveloping vertebral skeleton mirrors the anatomical features of the enclosed neural contents. During the cranio-caudally directed growth, spurts of elongation of the vertebral column may be too rapid for the slower growth rate of the spinal cord and nerve roots. The resulting disproportion of growth between spine and nervous system is compensated for by adaptive scoliotic curvature of the otherwise normally growing spine. The proposed pathogenetic concept readily explains the main clinical features of the deformity and is supported by a spring model experiment. [13] Roth, M. (1981), Idiopathic scoliosis and Scheuermann “disease”: Essentially identical manifestation of neuron-vertebral growth disproportion. Radiol. Diagn. 22 (1981), H.3, 380-391 Original language: German Abstract: With the knowledge postulated on the disproportionate growth between the nervous tissue and the spine, Roth gives with his spring models a true to nature morphological presentation of the proportioned relations of the cord and the deformed vertebrae (compression of the spring windings at the concavity, rope in the concavity). As the eccentric position of the cord and cauda was already known in literature (Lindgren 1941, Jirout 1964) Roth reverses this with arguments as it was seen as a secondary effect of the deformity towards a primary position with the deformity of the spine as a nervous tissue conducted developmental feature of the surrounding bony tissue. To Roth` s finding the lower thoracic and thoracolumbar spine function as the most predilected area for the first dicongruencies between the two types of growth in otherwise healthy children in their growth. In his

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view the modificated muscular activity to keep the new posture with its relocated axial loading and unloading. He also gives a understandable discussion in his concept of the existence of congenital scoliosis and kyphosis. [14] Roth, M. (1985), Once more spinal cord and scoliosis, Acta Chir. Orthop. Traum. Czech. 52, 1985, no. 6, p. 532-543 Original Language: Czech Abstract: Morphogenesis of the spine as well as of the neurocranium cannot be understood from the growing bone tissue alone, regardless of the morphology and growth peculiarities of the neural content, the brain and the spinal cord-nerve roots complex. Idiopathic scoliosis may be explained as a consequence of excessive discrepancy between the neural and the vertebral growth rates. Growth rate differential is a well-established factor of morphogenesis resulting, among others, in curvature of two adjacent structures growing in length at different rates. The periods of growth spurt are particularly prone to neurovertebral growth disproportionateness since the spinal cord-nerve roots complex may be unable to keep pace with the too rapidly growing spine. The latter is then laid in adaptive kyphoscoliotic curvatures alone the growth insufficient neural content. The relative growth deficit of the “Wirbelbogenreihe”as compared with the “Wirbelkorperreihe”, a feature characteristic for idiopathic scoliosis seems to result from the primary retarding effect of the spinal nerves, which run reins like along the pedicles. This neural retarding effect may be elicited either by excessive stimulation of the vertebral growth or by inhibition of the spinal neural growth, or by combination of both. The basic gross features of idiopathic scoliosis including the deformity of the thoracic cage and the concave sided eccentric position of the spinal cord may be reproduced by means of a neuro-costovertebral model. [15] Roth, M. (1986), Cranio-cervical growth collision: another explanation of the Arnold Chiari malformation and of basilar impression, Neuroradiology (1986) 28: 187-194 Original language: English Abstract: Analysis of neuro-cranio-spinal development suggests a cranio-cervical growth conflict as the cause of the Arnold Chiari malformation and of basilar impression .In gross (Meningomyelocele) and slight (adolescent deformities) impairment of distal spinal growth, a reversal of cervical growth occurs, the initial descent (uncoiling) of the primordial brain curvatures is compromised owing to the growth collision with the ascending cervical spine. The availability of space is subject of the struggle. [16] Roth, M (1989’, The `Enveloping` versus the Biomechanical function of the spine, Cs. Radiologie 43, 1989, No. 1, c. 1-13. Original language: Czech Abstract: As a continuation of argumentation presented in a number of previous communications, the author advocates the view according to which the developing spine and its neural content (“ spinal cord-nerve roots complex”) are linked by an equally intimate morphogenetic relation like that existing between the brain and its skeletogenic envelope. A specific feature of the neurovertebral developmental relation consists in the fact that the elongated spinal cord-nerve roots complex is enveloped by its skeletogenic case both in the transversal and in the longitudinal direction. The matter is further complicated by lagging of the spinal neural growth behind that of the vertebral column. The development of the basic anatomical features of the individual vertebrae such as their length and width, the girth of the vertebral body as well as the shape of the intervertebral foramina cannot be understood without taking into account the gross developmental dynamics of the two main components of the axial organ, viz., of the spinal cord-nerve roots complex and of the vertebral column. [17] Roth, M. (1994), Traumatic Spondodylolysis in the hedgehog. A contribution to the Problem of Dysplasia of the Isthmus, Z. Orthop. 132 (1994), p. 33-37 Original language: German Traumatic Spondylolysis in a hedgehog is reported. On the basis of that rare thinning of the vertebral isthmus frequently associated with Spondylolysis in man is claimed to be related to the “neuroenveloping “function of the spine shared with that of the neurocranium. Dysplasia of the isthmus results from abnormal ganglio-foraminal interrelation in the embryo rather than from any primary derangement of the vertebral bone growth proper.

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Acknowledgments We thank Milan Roth’s widow Mrs. Milada Rothova and sons Tomas and Michal for their help in providing his biographical material and photographs and their approval to use all his published work to be brought under attention of the present medical world interested in unanswered questions on growth. Also thanks to Dr.Kayo Styblo PhD, now orthopedic surgeon in Zaandam the Netherlands. who originates from Czechoslovakia, translated the Czech articles of Milan Roth into Dutch.

Chapter 4 Etiology

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Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved. doi:10.3233/978-1-58603-888-5-197

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Etiologic theories of idiopathic scoliosis: autonomic nervous system and the leptinsympathetic nervous system concept for the pathogenesis of adolescent idiopathic scoliosis RG BURWELL1, PH DANGERFIELD2, A MOULTON3, SI ANDERSON4 1

Centre for Spinal Studies and Surgery, Nottingham University Hospitals Trust, Queen’s Medical Centre Campus, Nottingham, UK, 2 The University of Liverpool, The Royal Liverpool Children’s Hospital and Staffordshire University, 3Department of Orthopaedic Surgery, King’s Mill Hospital, Mansfield, UK, and 4School of Biomedical Sciences, University of Nottingham, UK Abstract. The autonomic nervous system through its hypothalamic neuroendocrine control of puberty, skeletal growth and menarche contributes importantly to the pathogenesis of adolescent idiopathic scoliosis (AIS). Melatonin dysfunction detected in AIS subjects also involves the autonomic nervous system. The thoracospinal concept for the pathogenesis of right thoracic AIS in girls thought by some to result from dysfunction of the sympathetic nervous system (SNS), is supported by recent vascular and peripheral nerve studies. Lower body mass index (BMI).in girls with AIS is associated with decreased circulating leptin levels. Leptin, secreted by adipocytes, is a master hormone with many regulatory functions for growth and reproduction, including: 1) appetite repression, anorexigenic; 2) initiation of puberty in girls in a permissive action, and 3) in mice, longitudinal bone growth, chondrogenic and angiogenic, and in bone formation, antiosteogenic acting centrally through the SNS and possibly directly. In AIS girls, autonomic nervous system activity was reported to be higher than in controls. We suggest that in AIS susceptible girls, given adequate nutrition and energy stores, circulating leptin talks to the hypothalamus where dysfunction leads to an altered sensitivity to leptin resulting in increased SNS activity contributing with neuroendocrine mechanisms to: 1) earlier age at, and increased peak height velocity, 2) general skeletal overgrowth, 3) earlier skeletal maturation, 4) extraspinal skeletal length asymmetries, including periapical ribs and ilia, 5) generalized osteopenia, and 6) lower BMI. The SNS-driven effects may also add adventitious changes to the spine including asymmetries complicating the neuroendocrine effects on adolescent spinal growth. In AIS pathogenesis, the leptin-SNS concept is complementary to our NOTOM escalator concept involving the somatic nervous system. Together these two concepts view AIS in girls as being initiated by a hypothalamic dysfunction of energy metabolism (bioenergetics) affecting skeletal growth in the trunk. Where, in susceptible girls, the postural mechanisms of the somatic nervous system fail to control the asymmetric spinal and/or rib growth changes in a rapidly enlarging adolescent spine; this failure becomes evident as mild backshape shape asymmetry, or scoliosis. The environmentally-enhanced stature of normal subjects in the last 300 years, in girls susceptible to AIS, may have exaggerated any

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R.G. Burwell et al. / Etiologic Theories of Idiopathic Scoliosis developmental dysharmony between the autonomic and somatic nervous systems being fought out in the spine and trunk of the girl – possibly making mild back-shape asymmetry, or scoliosis more prevalent today than hitherto. Key words. Idiopathic scoliosis, pathogenesis, spine, leptin, hypothalamus, sympathetic nervous system

1. Introduction 1.1. Autonomic nervous system and the new neuroskeletal biology In reviewing the new field of neuroskeletal biology Patel and Elefteriou [1] summarize long-standing clinical observations relating to bone and the nervous system including reflex sympathetic dystrophy, hyperplastic callus associated with head injury and myelomeningocoele, and osteopenia associated with stroke, spinal cord injury and peripheral neuropathy. They comment that the hypothalamus with its ‘sensing’ functions and capacity to send efferent hormonal and neuronal signals has the capacity to integrate skeletal physiology with the daily physiological changes triggered by the environment and by endogenous rhythms. Conflicting reports on the effect of βadrenergic blockers for risk of fractures are published [2-4]. The new field of neuroskeletal biology involves leptin, the hypothalamus, sympathetic nervous system and skeletal tissues [1]. Here, we consider a theoretical appraisal of how this new field of neuroskeletal biology may relate to growing skeletal tissues and AIS pathogenesis (Figure 1). 1.2 The leptin-hypothalamic-SNS concept (leptin-SNS concept, Figure) We suggest that in AIS susceptible girls, given adequate nutrition and energy stores, circulating leptin talks to the hypothalamus where dysfunction leads to an altered sensitivity to leptin resulting in increased sympathetic nervous system (SNS) activity contributing to skeletal changes (mainly in the trunk and upper arms) including putative asymmetric changes to the spine complicating the traditional neuroendocrine effects on spinal growth. In AIS pathogenesis. the leptin-SNS concept is complementary to our NOTOM escalator concept involving the somatic nervous system [14,15]. Together these two concepts view AIS in girls as being initiated by a hypothalamic dysfunction of energy metabolism (bioenergetics) favouring growth and where, in susceptible subjects, the postural mechanisms of the somatic nervous system fail to control the asymmetric spinal changes in a rapidly enlarging adolescent spine; this failure becomes evident as mild back-shape shape asymmetry, or scoliosis deformity. In this paper 1) we review evidence on which the leptin-SNS concept for AIS pathogenesis is based, and 2) outline the hypothesis. 1.3. Autonomic nervous system and AIS 1.3.1. Menarche In AIS, the age at menarche has been reported as early, normal and delayed [5].

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Figure 1. Concepts for the pathogenesis of AIS involving the autonomic and somatic nervous systems. The sensory input, motor output and PPC relate to the somatic nervous system and the rest to the autonomic nervous system. Shown are some neuroendocrine controls (‘infantry’) and sympathetic nervous system (‘cavalry’) of skeletal and adipose tissues. Ganglia=ganglionated sympathetic trunk. Sympathetic nerves are shown as thin continuous lines and hormones as interrupted lines. Pre- and post-ganglionic sympathetic nerves are shown on one side with arrows indicating enhancement of function and blunted lines as inhibition. F=frontal lobe, P=parietal lobe, PPC=posterior parietal cortex (Area 7 with body schema), VC=visual cortex, GnRH=gonadotropin-releasing hormone, FSH=follicle stimulating hormone, LH=luteinizing hormone, ICA=intercostal artery,., T/L=thoracolumbar. For further details of the somatic nervous system concept see [13-15].

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1.3.2. Skeletal growth velocity An undisputed factor in AIS curve progression is pubertal skeletal growth and its velocity as it affects the spine [5]. In AIS girls, greater peak height velocity of growth in early puberty may be related to the increased secretion of growth hormone [7-9] dehydroepiandrosterone (DHA) and plasma testosterone [7,10,11] with normal estrogens [10,11]. The pathogenesis of AIS in girls evidently involves the autonomic nervous system and neurendocrine mechanisms. The mechanism(s) by which velocity of skeletal growth contributes to the pathogenesis of AIS in girls is unclear [12]. Castelein [12] states that the role of growth in the pathogenesis of AIS may be causative, conditional, amplifying, or coincidental. We suggest [13-15] that the dependence of AIS progression on growth is explained by rapid skeletal enlargement beyond the capacity of the postural mechanisms of the somatic nervous system to control the initiating deformity rather than by the velocity of growth, i.e. the role of skeletal growth is conditional [12]. 1.3.3. Circulating melatonin and melatonin-signaling pathway dysfunction In AIS subjects, a deficiency of circulating melatonin [16] and a systemic melatoninsignaling pathway dysfunction affecting osteoblasts and other cells [17] have been reported. The latter may represent a dysfunction of circadian rhythms in peripheral tissues similar to that detected in skin fibroblasts [18]. Any deficiency of circulating melatonin in AIS subjects could reflect altered sympathetic activity since the pineal gland is controlled by sympathetic fibers from the superior cervical ganglion – though normally one would expect any increased sympathetic activity to be reflected in greater nocturnal secretion of melatonin from the hypothalamus [Ebling FJP personal communication]. 1.3.4. Generalized osteopenia In girls with AIS, Cheung et al [19] found that the lower bone mass compared with controls could be explained by faster anthropometric growth, higher bone turnover, and lower calcium intake. The calcium intake in AIS patients was found to be very low with low consumption of dairy products, and likely to be insufficient for normal bone mineralization. Suh et al [20] reported evidence implicating imbalance and disturbed interaction of RANK ligand (RANKL) and the osteoprotegerin (OPG) decoy receptor in the osteopenia of AIS girls. 1.3.5. Disordered eating behavior There are several reports of lower body mass index in girls with AIS [5,19,21-23] but not all [24,25] and there is some evidence of disordered eating behavior [21,26,27]. The low body-mass index of girls with AIS is said not to be the result of the eating disorder [26]. 1.3.6. Right thoracic AIS in girls, sympathetic nervous system and rib-length asymmetry Sevastik’s thoracospinal concept [28-30] based on experimental, anatomical and clinical evidence, applies only to right thoracic adolescent idiopathic scoliosis in girls. There are six sequential processes as follows (linear causality): 1) Dysfunction of the autonomic nervous system[30]. 2) Increased vascularity of left anterior hemithorax. 3) Overgrowth of left periapical ribs which -

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4) disturbs the equilibrium of the forces that determine the normal alignment of the thoracic spine, that – 5) triggers the thoracospinal deformity simultaneously in the three cardinal planes. 6) Biomechanical spinal growth modulation [31]. This concept is supported by recent vascular [32] and peripheral nerve [33] studies 1.3.7. Leptin and AIS in girls In AIS girls, Qiu et al [23] reported a marked decrease in circulating leptin compared with controls. Positive correlations were found between leptin and each of age, menstrual status, weight, corrected height, body-mass index (BMI), Risser sign, bone mineral content and bone mineral density (lumbar spine and femoral neck) but not Cobb angle. Qiu et al comment on evidence strongly suggesting that leptin may 1) act on bone tissue through both central and peripheral pathways; a central pathway mediated by hypothalamic nuclei and the sympathetic nervous system (SNS), and peripherally “…by stimulating osteoblastic differentiation and inhibiting osteoclastic activity…” resulting in enhanced bone formation and reduced bone resorption; 2) regulate body growth during childhood and adolescence; and 3) mediate energy expenditure in females. 1.3.8. Body-mass index (BMI) and skeletal growth in the trunk of AIS and normal girls Elsewhere [25] we present a preliminary report showing in three groups of adolescent girls – normals, screened and preoperative - that body-mass index above and below mean levels separates subjects in all three groups with larger and smaller skeletal growth mainly in trunk width. It is hypothesized that: 1) normal trunk widening of the thoracic cage by hormones in human adolescence is supplemented via the sympathetic nervous system under leptinhypothalamic control influenced by energy stores (metabolic fuel); and 2) hypothalamic dysfunction with altered hypothalamic sensitivity to leptin through a SNS-driven asymmetric effect may create skeletal length asymmetries in vertebrae, ribs, upper arms and ilia, and initiate AIS. 1.3.9. Autonomic nervous system activity in AIS girls In a study of 56 AIS patients compared with 25 controls, a higher level of autonomic nervous system activity was found [34]. The authors suggested that this finding may be another manifestation of a systemic neurologic imbalance in AIS subjects. Cutaneous patterns of sympathetic activity are found in association with other clinical abnormalities of the musculoskeletal system [35]. 1.4. Hypothalamus (Figure) Buijs et al [36] write: The hypothalamus integrates information from the brain and body; this activity is essential for the survival of the individual (adaptation to the environment) and the species (reproduction). As a result, countless functions are regulated by the neuroendocrine and autonomic hypothalamic processes in concert with the appropriate behavior that is mediated by neuronal influences on other brain areas.

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1.5. Energy balance and signals in relation to growth and reproduction Energy balance requires precise coordination between peripheral nutrient-sensing molecules (related to food intake and stores) and central regulatory networks [37-42]. Fertility is gated by nutrition and the availability of energy reserves (metabolic fuel) stored as fat and glycogen to allow for growth and reproduction [41,42]. So that the normal female is ready for pregnancy and lactation, there is a link between body fat and the timing of puberty with leptin playing a permissive role [43] and kisspeptin activation of the G-protein coupled receptor-54 [41,44] providing a critical metabolic signal initiating puberty through pulsatile GnRH secretion [45] (Figure1). Other hypothalamic gates, or stiles, may have to be opened before menarche is reached [39]. 1.6. Leptin and the sympathetic nervous system – the new field of neuroskeletal biology Leptin, best known as a signal of energy sufficiency, is one of several hormones secreted by adipocytes. In girls there are gradual age- and body-weight related increases in circulating leptin levels [46]. Lower leptin levels, found in boys than girls [46], also rise until the early stages of puberty after which they decline [47]; leptin levels in men are lower than in women at all decades of life [40]. The sex difference may result from stimulatory effects of estrogen on leptin production and inhibitory actions of testosterone [47]. Leptin is a master pleiotropic hormone involved in the regulation of a variety of physiological processes, including [41,48]: 1) appetite repression, anorexigenic; 2) thermogenesis, 3) metabolism of glucose and lipid, 4) initiation of puberty in girls with kisspeptin stimulating GnRH secretion; and 5) complex effects on growing bones with fat having a protective effect on bone tissue [49]. Leptin is reported to stimulate linear growth by regulating the energy balance of the body and by stimulating the production and secretion of growth hormone; at the same time it has a direct effect on chondrocytes of the growth plate and is involved in bone remodelling [48]. Iwaniec et al [50] propose the hypothesis that hypothalamic leptin plays a role in coupling energy homeostasis and bone growth. Leptin appeared in evolution with the bony skeleton [51]. The coupling of skeletal growth to energy balance [48,50] involves leptin and Y2receptors on neuropeptide Y (NPY-ergic) hypothalamic neurons [52,53]. Other metabolic hormones, insulin and thyroid hormone as well as sex steroids also target hypothalamic neurons (arcuate nuclei) to ensure metabolic efficiency [41](Figure1). In mice, adipocytes talk to bone through leptin acting centrally through a hypothalamic pathway via the SNS [54,55] which releases the neurotransmitter noradrenaline to increase longitudinal bone growth and inhibit osteoblast function and proliferation [1,47,50,52]. In mice, leptin stimulates longitudinal bone growth, being chondro-osteogenic and angiogenic, and in bone formation, antiosteogenic acting centrally through the SNS and possibly with an opposite direct effect on bone [47,50,52]. The skeleton exerts an endocrine regulation of energy metabolism. [51,56] (Figure 1). 1.7. Sympathetic nervous system (SNS) and innervation of white adipose tissue White adipose tissue in rats and Siberian hamsters is innervated by the SNS [39,57,58] which may play a significant role in lipid mobilization [39,57], and inhibition of proliferation [57](Figure).

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2. Hypotheses 2.1. Leptin-SNS concept - effects on skeletal and white adipose tissues in girls with AIS In AIS susceptible girls, given adequate nutrition and energy stores, in the leptin-SNS concept circulating leptin talks to the hypothalamus with dysfunction and altered sensitivity to leptin which leads to increased SNS activity contributing with neuroendocrine mechanisms to (Figure 1): a) earlier age at, and increased peak height velocity [5,9,59] b) general skeletal overgrowth [5,60] c) earlier skeletal maturation [5,25] d) extra-spinal skeletal length asymmetries, including periapical ribs [5,13,14] and ilia [61] e) generalized osteopenia [19,21], and f) lower body fat and BMI [5,19,21-23]. The extra-spinal skeletal length asymmetries are a manifestation of developmental stress in hypothalamic neurons as they adjust to lower leptin levels which they helped to create. The SNS-driven effects also add adventitious changes to the spine including asymmetries complicating the neuroendocrine effects on spinal growth. The SNS contributes to the osteopenia of AIS by activating β2-adrenergic receptors on osteoblasts [62] causing them to produce RANKL which acts on the osteoclast receptor RANK to stimulate osteoclast formation [63]. In young ballet dancers, the high prevalence of scoliosis, fractures and delayed menarche attributed to hypoestrogenism [64] may also involve low circulating leptin levels and increased hypothalamic-SNS-driven activity on growing bones. 2.2. Putative changes in the spine and the somatic nervous system in AIS When the SNS-driven putative adventitious changes in the enlarging adolescent spine are associated with relative postural maturational delay in the somatic nervous system the spine deforms [13-15]. The postural mechanism may be linked to higher testosterone levels in AIS girls affecting brain white matter maturation in girls [7,10,11,15]. There is direct and indirect evidence suggesting that asymmetries affect vertebral body growth plates [13,14,65]. 2.3. Putative hypothalamic dysfunction and the NOTOM escalator concept In AIS girls, we hypothesize that the hypothalamic gates controlling skeletal growth via neuroendocrine and autonomic outputs open earlier to advance skeletal maturation [Ebling FJP, personal communication]. The leptin effect via the SNS may add to the neuroendocrine effect of both growth hormone and estrogen driving adolescent skeletal growth in girls [5-8]. Alternatively, the dysfunction in hypothalamic neurons as putative altered sensitivity to leptin may result from an abnormality of a Gprotein-coupled receptor to leptin, or Gprotein - possibly as the critical part of a systemic abnormality similar to the melatonin-signalling pathway dysfunction detected in osteoblasts, muscle cells and lymphocytes [66]. In AIS pathogenesis, the leptin-SNS concept is complementary to our NOTOM escalator concept involving the somatic nervous system [14,15]. Together these two

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concepts enable AIS to be viewed as being initiated in girls by a hypothalamic dysfunction of energy metabolism (bioenergetics) favoring growth in a secular trend; and where, in susceptible subjects, the postural mechanisms of the somatic nervous system fail to control the adventitious spinal changes in a rapidly enlarging adolescent spine; this failure becomes evident as mild back-shape shape asymmetry, or scoliosis deformity. 2.4. A secular trend in normal stature with significance for AIS pathogenesis? Stature has increased since the mid 18th century due to environmentally-induced changes [67,68]. This secular height increase, attributed to better nutrition and reduced infections by advances in technology resulting in vital organs having improving robustness and capacity, is interpreted by the hypothesis of technophysio evolution [69]. The biological mechanism underlying this secular trend in stature like the declining age at puberty [42] is likely to be driven by the hypothalamus [Ebling FJP, personal communication]. We suggest that in those girls who develop AIS the increased size for age from these secular changes in spine and trunk may be a factor weakening control by postural mechanisms for holding a spine straight. This environmentally-enhanced stature of normal subjects in the last 300 years in girls susceptible to AIS may have exaggerated any developmental dysharmony between the autonomic and somatic nervous systems being fought out in the spine and trunk of the girl – possibly making mild back-shape asymmetry, or scoliosis more prevalent today than hitherto. Acknowledgement We thank Professor FJP Ebling for a discussion of the autonomic nervous system and AIS and subsequently for reading the text and making helpful comments. References [1] [2]

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Chapter 5 Screening and Prevention of Scoliosis

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Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved. doi:10.3233/978-1-58603-888-5-221

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Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved. doi:10.3233/978-1-58603-888-5-231

231

Positional relationship between leg rotation and lumbar spine during quiet standing N PARKER1, A GREENHALGH1,2, N CHOCKALINGAM1, P H DANGERFIELD1,3 1

Staffordshire University, Stoke on Trent ST4 2DF, 2University of Central Lancashire 3 The University of Liverpool Abstract. Healthcare professionals frequently evaluate spinal posture on visual assessment during the clinical examination. While this visual assessment of the spine has been shown to be unreliable, the use of a plumbline as to aid clinical visual assessment has also been reported. There is a "normal" sagittal contour that functions quite well in healthy people. It positions the head in space, it protects the neural axis, and it allows efficient, pain-free motion. Lumbar lordosis is routinely evaluated in most spine patients, but what constitutes a normal sagittal contour is less well defined. A key component of normal sagittal contour is lumbar lordosis. Changes in the lumbar lordosis frequently occur in pathological gait, usually in association with alterations in pelvic tilt, and commonly as a compensation for a limited range of flexion/extension at the hip joint. Recent investigations looked at the effect of hyperpronation on pelvic alignment in a standing position and supported the existence of a kinematic chain in healthy subjects, where hyperpronation can lead to an immediate shank and thigh internal rotation and change in pelvic position. While there is a wealth of research is available on the effectiveness of functional foot orthoses, the present investigation reports the effect of pronated foot position on the lumbar region of the back by employing an optoelectronic movement analysis system. Keywords: Lordosis, Lumbar, Spine, leg, foot, calcaneus

1.

Introduction

Lower back pain is a disabling condition that affects both the general and working population. While, Burton et al [1] indicated that shoe insoles are not recommended in the prevention of back problems, previous investigations have looked at the effectiveness of shoe inserts on back pain. The rationale for the use of foot orthoses to prevent or modify back pain is based on their impact attenuation ability during walking [2]. However, Dananberg [3, 4] has provided some evidence for an alternative rationale for treatment of back pain with functional foot orthoses. There is a "normal" sagittal contour of the spine that functions effectively in healthy people. It protects the neutral spine axis, and precipitates efficient, pain-free motion. Lumbar lordosis (LL) is a key component of the normal sagittal contour, it is routinely evaluated in most spine patients, but is not well defined. Changes in the LL frequently occur in pathological gait, usually in association with alterations in pelvic tilt, and commonly as a compensation for a limited range of flexion/extension at the hip joint [5, 6]. Although the invasiveness and risk of radiation preclude the use of the radiograph as

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a routine measurement in clinical practice, the gold standard measurement of LL is still a plain film x-ray [7,8,9,10]. Other spinal measurement techniques have been developed and reported [11], such as Goniometry [12], skin markers over the spinal processes [13] inclinometers [14] [15] and the use of flexible rulers [16]. These external measurements are easy to apply and non-invasive [17] [10]. Whittle & Levine [6] define the angle of LL as the difference in angle between the areas of skin over the T12-L1 junction and the L5-S1 junction, projected in the sagittal plane. Other researchers have reported different points of measurement relative to the technique being used [7, 8]. Whittle & Levine [6, 18-20] performed a range of studies to promote the use of gait analysis for measuring LL. They demonstrated a high level of significance and reliability when measuring LL based on extensive previous work using motion capture techniques. The relationship between the mechanics of the foot and lower extremity has been confirmed in various studies [21, 22] [23] [24, 25]. However the relationship between foot mechanics and the knee, hip and pelvis are less clear [26][27][28]. Studies investigating the lumbo-pelvic region demonstrated that increasing anterior pelvic tilt increased the LL and increasing posterior pelvic tilt did the opposite [18]. Later work showed the average pelvic tilt and average lumber lordosis relationship was out-of-phase. Pelvic tilt data was relatively consistent across subjects, but the LL showed a complete variety of patterns [6]. They concluded that LL has a highly individualist pattern of movement and that during gait appears to be variable from one subject to another which contrasted previous work which reported patterns of movement in the lumbar spine complemented those of the pelvis in the sagittal plane [29]. The maintenance of postural stability is a complex task that requires the coordination of visual, vestibular and somatosensory inputs [30]. Balance is defined as the process of maintaining the centre of gravity within the body’s base of support [31]. Centre of gravity (COG) is described by Winter [32]. Postural sway [33] compensates for small perturbations in stance; however the body cannot be considered as a complete segment. The trunk will always move independently to contribute to balancing the COM. Over time postural changes can occur due to muscular stretchweakness and weaknesses in proprioception. The current study investigates whether subtalar movement affects limb rotation and pelvic alignment, and if prevalent attempts to corrleate movement at the subtalar joints to movement and alterations in the lumbar lordosis.

2.

Materials and Methods

Ethical approval from appropriate ethics committee was received. Children and individuals receiving any type of treatment were excluded from the study. . Subjects were selected using convenience sampling according to their availability to data collection sessions. 17 subjects were recruited to the study (8 males and 9 females) between the ages of 18 and 40 (mean 26.23 years). Subjects were free of any musculoskeletal conditions and abnormalities which included back pain. All subjects gave an informed consent to take part in the study. Inclusion and exclusion criteria are detailed in Table 1.

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The upper age limit was set to 40 to exclude subjects with undetermined early degenerative changes in the lumbar spine. It has been suggested that the pathology peaks between the ages of 35 and 55 [34]. Subjects Inclusion criteria Exclusion criteria with obvious lower Ages between 18 and 40 Current or past back pain extremity deformity; No known lower extremity Obvious lower extremity particularly marked dysfunction dysfunction; particularly internal/external femoral marked internal/external or tibial torsion were femoral or tibial torsion. excluded on the basis that No LLD or LLD <5mm Leg Length Discrepancy these deformities may (>5mm) affect the translation of Palpable bony landmarks on the Subjects where bony landmarks rotation up the locomotor lumbar spine were not easily palpable. chain. Heavily pronated Foot Posture Index (Redmond Foot Posture score (Redmond et subjects were excluded et al 2006) score from -1 to +7 al 2006) of 7 or greater, <-1 from the study and only those with Foot Posture Table 1: Inclusion/Exclusion Index [35] score from -1 to +7 were admitted. Flexibility on the positive side of the score was permitted from subjects presenting easily palpable bony landmarks. Subjects with a leg length discrepancy >2cm were excluded in line with the European guidelines for the prevention of Low back pain [1]. Kinematic data was acquired using an optoelectronic motion analysis system (VICON, Oxford). The system utilised 8 infrared cameras to track 15mm reflective markers that were placed on specific landmarks on the lower limb, pelvic area and lumbar spine. The system was calibrated using the manufacturer’s guidelines at the beginning and end of each data collection session. Ground reaction forces (GRF) were collected using a force plate (AMTI). Pilot Study A single subject pilot study was completed and specific positions of the markers identified by the system were verified by using actual measurements. Two marker sets for evaluating lumbar spine curvature had previously been developed, 3Figure 1: 4 Marker Set (Whittle and Levine 1997) marker set and a 4-marker set [6]. The 4-marker set was chosen. Two standard VICON rigs were used each consisting of a flat baseplate, from the centre of which protruded a 100mm long wand with a retroflective marker mounted at its end. The marker on the upper baseplate was directly over L1 and the marker on the lower baseplate was directly over S2. The raw data was then exported to Microsoft Excel to calculate the magnitude of lumbar lordosis using a previously described method [6].

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Mean Age (years Weight (Kg) Height (m)

26.2 72.1 1.7

Standard Deviation ± 5.0 ± 11.4 ± 0.1

BMI (Kg/m2)

23.7

± 2.4

Range 21 - 36 53 - 97 1.6 1.87 19.2 28.4

Table 2: Participant characteristics n=17 (9 F: 8 M)

Standard anthropometric data (table 2) was collected and the Foot Posture Index (FPI) was determined by the same researcher (NP). Furthermore for every subject the skin markers for kinematic data acquisition were applied by the same researcher (NP). Prior to fixing the two rigs to the skin, the angle of the wand to the baseplate was validated on a supporting surface manually and by the system. The angle between the baseplate and the line joining the centres of the two markers was then calculated to be 42.84º. The subject was asked to stand in a relaxed position with their arms crossed and the palms of the hands resting on their shoulders to standardise the position and minimise any affect upper limb Test Conditions placement may have on the centre of pressure. Three Relaxed Standing dimensional kinematic data was collected for all conditions and 3.5° Rearfoot Wedges 3.5° Forefoot Wedges reported in table 3: 3.

3.5° Full foot Wedges 5° Rearfoot Wedges 5° Forefoot Wedges 5° Full foot Wedges 10° Rearfoot Wedges 10° Forefoot Wedges 10° Full foot Wedges

Results

Changes in magnitude of Lumbar Lordosis

Results for all subjects (Fig. 2) showed no substantial similarities between subjects. Table 3: Wedged conditions tested 105

100

Lumbar Lordosis (Degrees)

95

90

85

80

75

70 Standing

3º Rearfoot

3º Forefoot

3º Full

5º Rearfoot

Figure 2: Magnitude of Lumbar Lordosis

5º Forefoot

5º Full

10º Rearfoot 10º Forefoot

10º Full

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Analysing the change in LL from relaxed standing at rearfoot modes only shows a reduction that approaches a linear relationship (Figure 3).

Magnitude of Lumbar Lordosis (Degrees)

1.00

0.00

-1.00

-2.00

-3.00

-4.00

-5.00 3º Rearfoot

5º Rearfoot

10º Rearfoot

Figure 3: Change of Magnitude of Lordosis from relaxed standing for Rearfoot Conditions

Results were analysed according to Foot Posture Index (FPI) score. The objective of this exercise was to find if certain foot types affect the kinetic chain and therefore the magnitude of the LL in the standing position (Figure 4). The results suggest there may be a correlation between the slightly more pronated feet and an increased lumbar lordosis. There may also be correlations between the 3º and 5º rearfoot and forefoot wedge modes however the same cannot be said for the 10º results. Regarding the subjects with FPI scores of 2:2 there appears to be limited uniformity to the results, however all results except one are in the negative presenting a reduction in lordosis. 12.00 10.00

Magnitude of Lumbar Lordosis (Degrees)

8.00 6.00 4.00 2.00 0.00 -2.00 -4.00 -6.00 -8.00 -10.00 3º RF

3º FF

3º Full

5º RF

5º FF Total FPI 5:5

5º Full

10º RF

10º FF

10º Full

Total FPI 2:2

Figure 4: Subjects with Homogeneous FPI scores of 5:5 (n=4) and 2:2 (n=3)

Changes in the projection of the centre of gravity in the transverse plane (PCOG) Results from a sample of subjects were analysed to assess the translation of the centre of gravity. A negative change indicates that body posture had swayed anteriorly. The converse is true for a positive change in the COP.

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Mean values (Figure 6), show all points producing negative values, indicating an anterior translation of the centre of gravity. There is a wide variety of results with little pattern. Standard deviations show that this may not be the case for larger populations 12.00

Magnitude of Lumbar Lordosis (Degrees)

10.00

8.00

6.00

4.00

2.00

0.00 RF

FF 3º Results

5º Results

Figure 5: Subjects with Homogeneous FPI scores of 5:5 (Rearfoot and Forefoot only)

10.00

Translation of PCOG (mm)

5.00

0.00

-5.00

-10.00

-15.00 3º RF

3º FF

3º Full

5º RF

5º FF Mean

+2SD

5º Full

10º RF

10º FF

10º Full

-2SD

Figure 6: Mean Translation of Projection of COG (-ve = Ant Translation)

4.

Discussion

The main aims of the study were; to develop a measurement technique to show that LL can be measured successfully, via non-invasive motion capture analysis; to measure the relationship of the LL to foot function and to consider the effect of foot function on the centre of gravity of the body. The pilot study confirmed the work of previous studies [6, 18, 36]. In standing posture, increasing the amount of pelvic tilt increases the angle of lumbar lordosis, and vice versa. The intention of this research was to deliver normative data on the topic as a baseline before studying pathologic subjects. The direct foot to lordosis relationship was studied as previous investigations pertinent to

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237

other links had already been presented [23-27]. Post study evaluation was unable to elucidate trends from the findings. Changes in magnitude of Lumbar Lordosis Changes in LL showed no general patterns across the range of subjects. This agreed with previous research where an out-of-phase relationship between the average pelvic tilt and the average LL was found [20]. Different effects were found at the lumbar level depending where the wedge is placed, but with no noticeable patterns. Descriptive data from the present study supports the null hypothesis that, as wedges are applied to the rearfoot in increasing amounts the lordosis decreases (Fig. 3), but not with any direct relationship. Making comparisons across foot posture type there appeared to be a correlation between the slightly more pronated feet and an increased lumbar lordosis. The 3º and 5º rearfoot and forefoot wedge modes seemed to show a general increase, however the same cannot be said for the 10º results. This may be because the foot reached maximal pronation or calcaneal eversion and further eversion was not possible. By inference, and notwithstanding the small sample size, the question of whether such interventions can really alter anything at lumbopelvic level by altering foot posture remains. Changes in the projection of the centre of gravity in the transverse plane (PCOG) Hertel [37] postulated that the interface between differently shaped feet (as described by Root [38]) and a force plate could influence ground reaction forces and thus postural control measures during objective assessment of postural stability during single-leg stance. The purpose of this part of the study was to investigate if a foot more akin to pes planus posture affected postural stability by causing an anterior shift in the COP. There was a general movement of the PCOG in the anterior direction (Figure 6), suggesting that foot pronation induced a forward movement of the PCOG across the small sample size. The standard deviations show that this might not be the case for a larger population. If the body maintains balance in response to an anterior shift in the PCOG by altering the upper limb to lower limb relationship, then it did not show in the magnitude of the LL results and may take place elsewhere. Results of the present study suggest excessively pronated feet may cause an anterior translation of the PCOG although the findings are not supported in the literature. This reinforces the common anecdotal belief that pronated feet alter body posture in the anterior/posterior direction. Unfortunately no clear pattern evolved from the results and a larger sample size is recommended for future studies. Correction of excessive pronation may help to prevent the change in PCOG, although this study didn’t demonstrate a link to lumbar posture. The results do not support the research aim which was to find whether subtalar movement affects limb rotation and pelvic alignment, and if prevalent attempts to corrleate movement at the subtalar joints to movement and alterations in the lumbar lordosis. 5. Limitations The primary limitation of this study is the small sample size. A larger population of subjects would have contributed more validity to the results. Tighter Inclusion criteria would have made demonstrating trends in the results more achievable. Direct comparison of the results with previous studies was not possible due to the different methodology accompanied with the innovative parameters being measured.

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Reproducing previous studies [25] to validate their results could have been incorporated into the study methodology to allow direct comparisons with previously reported research (REFERENCES). Using smaller wedges may have limited the study to very small fluctuations in lumbar posture in the test group whereas larger wedges, although not representative of the clinical situation may have produced better results. The results of this study were dependent on visualizing changes within a static model, with the lack of conclusive results from this study, further investigation where dynamic stresses are in place and can be assessed, should be considered. 6. Conclusion The response of the Lumbar lordosis to changes in foot posture is varied and the paucity of relationships suggest further studies are required. There may be merit in investigating subjects with different back postures to establish a degenerative link. The results suggest that pronated feet cause an anterior movement of the centre of gravity and reinforce the common anecdotal belief that pronated feet alter body position and posture, although compensation for these changes may not be taking place exclusively in the lordotic region. References [1] [2] [3] [4] [5] [6] [7] [8] [9] [10] [11] [12] [13] [14] [15] [16] [17]

[18] [19]

Burton, A.K., et al., European guidelines for prevention in low back pain, in European Spine Journal. 2006. S136-S168. Wosk, J. and A.S. Voloshin, Low back pain: conservative treatment with artificial shock absorbers, in Arch Phys Med Rehabil. 1985. p. 145--148. Dananberg, H.J., Gait style as an etiology to chronic postural pain. Part II. Postural compensatory process, in J Am Podiatr Med Assoc. 1993. p. 615--624. Dananberg, H.J., Gait style as an etiology to chronic postural pain. Part I. Functional hallux limitus, in J Am Podiatr Med Assoc. 1993. p. 433--441. 5Whittle, M., Gait Analysis:An Introduction. 1991. Whittle and D. Levine, Measurement of lumbar lordosis as a component of clinical gait analysis, in Gait and Posture. 1997. p. 101-107. Polly, D.W., et al., Measurement of lumbar lordosis. Evaluation of intraobserver, interobserver, and technique variability, in Spine. 1996. p. 1530--5; discussion 1535-6. Chernukha, K.V., R.H. Daffner, and D.H. Reigel, Lumbar lordosis measurement. A new method versus Cobb technique, in Spine. 1998. p. 74--9; discussion 79-80. Chen, Y.L., Vertebral centroid measurement of lumbar lordosis compared with the Cobb technique, in Spine. 1999. p. 1786--1790. Ng, J.K., et al., Range of motion and lordosis of the lumbar spine: reliability of measurement and normative values, in Spine. 2001. p. 53--60. Pearcy, M., Measurment of back and spinal mobility, in Clincal Biomechanics. 1986. p. 44-51. Burdett, R.G., K.E. Brown, and M.P. Fall, Reliability and validity of four instruments for measuring lumbar spine and pelvic positions, in Phys Ther. 1986. p. 677--684. Hart, D. and S. Rose, reliability of a non-invasive method for measuring lumbar curve, in Clinical Biomechanics. 1986. p. 44-51. Rondinelli, R., et al., Estimation of normal lumbar flexion with surface inclinometry. A comparison of three methods, in Am J Phys Med Rehabil. 1992. p. 219--224. Williams, R., et al., Reliability of the modified-modified Schöber and double inclinometer methods for measuring lumbar flexion and extension, in Phys Ther. 1993. p. 33--44. Lovell, F.W., J.M. Rothstein, and W.J. Personius, Reliability of clinical measurements of lumbar lordosis taken with a flexible rule, in Phys Ther. 1989. p. 96--105. Watson, A.W. and C. McDonncha, A reliable technique for the assessment of posture: assessment criteria for aspects of posture, in Journal of Sports Medicine and Physical Fitness [NLM - MEDLINE]. 2000. p. 260--. Levine, D. and M.W. Whittle, The effects of pelvic movement on lumbar lordosis in the standing position, in J Orthop Sports Phys Ther. 1996. p. 130--135. Whittle, Three-dimensional motion of the center of gravity of the body during walking, in Human Movement Science. 1997. p. 347--355.

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[20] Whittle, M. and D. Levine, Three-dimensional relationships between the movements of the pelvis and lumbar spine during normal gait, in Human Movement Science. 1999. p. 681-692. [21] Perry, J., Gait Analysis:Normal and Pathological Function. 1992. [22] Perry, J., Anatomy and biomechanics of the hindfoot, in Clin Orthop Relat Res. 1983. p. 9--15. [23] Nigg, B.M., G.K. Cole, and W. Nachbauer, Effects of arch height of the foot on angular motion of the lower extremities in running, in J Biomech. 1993. p. 909--916. [24] Nawoczenski, D.A., C.L. Saltzman, and T.M. Cook, The effect of foot structure on the threedimensional kinematic coupling behavior of the leg and rear foot, in Physical Therapy. 1998. p. 404--. [25] Khamis, S. and Z. Yizhar, Effect of feet hyperpronation on pelvic alignment in a standing position, in Gait Posture. 2006. [26] Reischl, S.F., et al., Relationship between foot pronation and rotation of the tibia and femur during walking, in Foot Ankle Int. 1999. p. 513--520. [27] Nester, C., The relationship between transverse plane leg rotation and transverse plane motion at the knee and hip during normal walking, in Gait Posture. 2000. p. 251--256. [28] Powers, C.M., et al., Comparison of foot pronation and lower extremity rotation in persons with and without patellofemoral pain, in Foot Ankle Int. 2002. p. 634--640. [29] Crosbie, J., R. Vachalathiti, and R. Smith, Patterns of spinal motion during walking, in Gait and Posture. 1997. p. 6-12. [30] Lord, S.R. and H.B. Menz, Visual contributions to postural stability in older adults, in Gerontology. 2000. p. 306--310. [31] Cote, K.P., et al., Effects of Pronated and Supinated Foot Postures on Static and Dynamic Postural Stability, in J Athl Train. 2005. p. 41--46. [32] 3Winter, D.A., A.B.C. (anatomy, biomechanics and control) of balance during standing and walking. 1995: Waterloo, Ontario : Waterloo Biomechanics. [33] .Kirby KA, Foot and Lower Extremity Biomechanics II: Precision Intricast Newsletters, 1997-2002. 2002: Precision Intricast, Inc., Payson, AZ, , pp. [34] Andersson, G.B.J., The Adult Spine: Principles and Practice, Fryomoyer, Editor. 1997, LippencottRaven, Philidelphia. p. 93-141. [35] Redmond, A., J.Crosbie, and R.Ouvrier, Development and validation of a novel rating system for scoring foot posture: the Foot Posture Index, in Clinical Biomechanics. 2006. p. 89-98. [36] Day, J.W., G.L. Smidt, and T. Lehmann, Effect of pelvic tilt on standing posture, in Phys Ther. 1984. p. 510--516. [37] Hertel, J., M. Gay, and C. Denegar, Differences in Postural Control During Single-Leg Stance Among Healthy Individuals With Different Foot Types, in J Athl Train. 2002. p. 129--132. [38] Root, M., W. Orien, and J. Weed, Normal and abnormalfunction of the foot: Clinical biomechanics. 1977: Clinical Biomechanics Corp. Los Angeles.

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Aetiology of Idiopathic Scoliosis. What have we learned from School Screening? T B. GRIVAS, E S. VASILIADIS, G RODOPOULOS Scoliosis Clinic, Orthopaedic Department, “Thriasio” General Hospital, G. Genimata Avenue, Magula, 19600, Athens, Greece. [email protected] Abstract. The value of school screening as a research tool for Idiopathic Scoliosis (IS) aetiology has not been recognized adequately in the literature. The aim of the present study is to summarize the contribution of school screening in research of IS aetiology. All the relative publications about research of IS aetiology which originated from our scoliosis school screening program were analyzed. Information is provided about a) the influence of environmental factors in IS prevalence, b) the IS prevalence in girls with visual deficiency, c) the role of melatonin in IS pathogenesis, d) the age at menarche in IS girls and its relation to laterality of the curve, e) the role of the brain in trunk asymmetry and IS pathogenesis, f) the role of the thoracic cage in IS pathogenesis, g) the impact of the lateral spinal profile, h) the role of the intervertebral discs in IS pathogenesis, i) the association of cavus foot with IS and j) anthropometric data in IS patients. The present study provides evidence to support that school screening programs should be continued not only for early detection of IS or for health care purposes, but also as a basis for epidemiological surveys until we learn much more about the aetiology of IS. Keywords. Idiopathic scoliosis, school screening, aetiology of idiopathic scoliosis

1.

Introduction

Scoliosis school screening (SSS) continues to be controversial. There are studies which both support [1, 2] and discourage [3, 4] routine SSS. The initial goal of SSS is to detect scoliosis at an early stage when deformity is likely to go unnoticed [5] and a less invasive treatment is more effective [6], although it is not a diagnostic process. Negativists to SSS programs have focused on concerns about a low predictive value of screening and the cost effectiveness of referral. There have also been concerns about the possibility of unnecessary treatment including brace use and the effect of exposure to radiation when radiographs are obtained [5, 7, 8, 9]. On the contrary there is evidence that SSS provide valuable data for research on IS aetiology [10, 11, 12, 13, 14, 15]. Even the opponents of SSS agree that it has a role and should be carried out as a basis for epidemiological surveys until we know much more about the aetiology and factors likely to determine the natural history of late-onset IS [16]. The present study highlights the input of our SSS program in research of IS aetiology.

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2.

241

Material and Methods

We reviewed all the relative publications which originated from the “Thriasio” SSS program [17] and provided some input in research of IS aetiology. 3.

Results

IS prevalence at “Thriasio Pedio”, which is a heavily industrialized area was found similar to the prevalence reported in other non-industrialized areas of Greece. This implies that industrial environmental factors do not play any role for a possible difference in IS prevalence [18]. IS prevalence has been reported to be different in various geographic latitudes and demonstrates higher values in northern countries. This observation could be related to the influence of the geography of a specific region on human biology and could be determined by socioeconomic and environmental factors such as temperature, humidity and light. [19] Furthermore, in a different study the regression of prevalence of IS and age at menarche by geographical latitude was found statistically significant (p < 0.001) and both they were following a parallel course of their regression curves, especially in latitudes northern than 25 degrees, which means that late age at menarche was parallel with higher prevalence of IS. The positive association between prevalence of IS and geographic latitude is present only in girls and not in boys. We hypothesized that the increased levels of melatonin in northern countries with poor light environmental conditions are reducing the secretion of LH and causes delayed age at menarche. Delayed puberty results in a prolonged period of spine vulnerability when other aetiological factors are contributing to the development of IS [20]. Interestingly, in blind girls who were found to have a delayed age at menarche, IS prevalence was found to be 42.3%! [21, 22]. An aetiopathogenic role of cerebral cortex function in the determination of the thoracic surface morphology of the trunk was hypothesized after finding a possitive correlation between trunk asymmetry and lateralization of the brain as expressed by handedness in in children who are entitled at risk to develope IS. [23, 24] In a study of rib vertebra angles (RVAs) we showed that scoliotic children with small curves had under developed thoracic cage compared to non scoliotic counterparts. It was suggested that the differences of the RVAs between right and left side were the expression of asymmetric muscle forces acting on the thoracic cage, which deforms early and possibly transfers the deforming forces to the spine [13]. In a different study no correlation was found between the Cobb angle and the “rib index” [12] of thoracic, thoracolumbar and lumbar curves. A positive correlation of the “rib index” with each of T2, T3, T4, T5, T6 and T7 Lateral Spinal Profile (LSP) in girls with lumbar curves was found. The mean age of non-scoliotics was 1.5-2 years younger than scoliotics. Rib index was approximately 1.5, which implies pronounced thoracic asymmetry in terms of rib hump, in all SSS referrals who presented with an already developed deformity of the thorax. 70% of them were scoliotics, 10% had a curve < 9o and 20% had straight spines, as confirmed by an x-ray. This observation supports the hypothesis that in idiopathic scoliosis the deformity of the thorax precedes the deformity of the spine [12, 25]. Furthermore, the correlation between the rib cage

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deformity and the deformity of the spine is weak in younger children, [25] as 25% of children with ATI ≥7º had a spinal curve under 10º or had a straight spine. As a result of the effect of growth on the correlation between the thoracic surface deformity and the spinal deformity, the predictive value of the existing formulas which calculate the Cobb angle from surface measurements is poor. Therefore our recommendation is to take into consideration the effect of growth when developing such predictive models, otherwise they can be inaccurate. In a study of the LSP of mild (10o-20o) scoliotic curves, we demonstrated that hypokyphosis of the thoracic spine by alleviating axial rotation is not a predisposing factor but rather a compensatory mechanism for the initiation of mild scoliotic curves [14]. In a radiological study of SSS referrals, we showed that in mild scoliotic curves, the deformity appears first at the level of the intervertebral disc (IVD), which is found wedged. The deformity of the vertebral body or of the spinal column follows. The eccentric intervertebral disc in the scoliotic spine, through variation in its water concentration, produces asymmetrically cyclical load during the 24-hour period and an asymmetrical growth of the vertebral body (Hueter-Volkman’s law). The deformation of the apical IVD seems to be an important progressive factor in IS pathogenesis [26]. Flat foot, as well as cavus foot which was previously reported that is correlated positively with IS was not confirmed in our study [10]. A variety of findings regarding the stature and weight of IS children has been published. In a study in Mediterranean population, the somatometric parameters of height and weight in children with scoliosis, regardless of curve type and site, are not statistically different from their non scoliotic counterparts [15]. Finally, studying the age at menarche in IS girls it was found that there was not statistically significant difference of this variable between scoliotic and non scoliotic girls in the Mediterranean population studied [11]. 4.

Discussion

The important findings on the lack of statistically significant difference of the age at menarche between scoliotic and non scoliotic girls in the studied Mediterranean population and the similar findings on the somatometric parameters of height and weight in children with scoliosis, which are not statistically different from their non scoliotic counterparts motivated us to explore the possible role and impact of the geography - environmental factors (the light) on the pathogenesis of scoliosis. The findings are reported in our results. These observations could not be figured out without running a SSS program. The early detection of IS has been a major and growing commitment of Orthopaedists since the early 1960s. Early detection implies early treatment and by that less surgery. Although SSS’s aim is prevention and it should be regarded as a criterion of welfare of our civilization, it has been criticized for its negative cost-effectiveness. It is clear that a realistic evaluation of the cost is not feasible and could result in inaccurate overestimation of the total cost as it might take into consideration many qualitative and subjective factors [17]. On the other hand the financial profit from research of IS aetiology, which was analyzed in the present study cannot be estimated.

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The present study provides evidence to support that school screening programs should be continued not only for early detection of IS but also as a basis for epidemiological surveys until we learn much more about the aetiology of IS. References [1] [2] [3] [4] [5] [6]

[7] [8] [9] [10]

[11]

[12] [13]

[14]

[15] [16] [17] [18]

[19] [20]

[21] [22] [23]

Ashworth MA, JA Hancock, L Ashworth, KA Tessier. Scoliosis screening. An approach to cost/benefit analysis. Spine 13(10) (1988) 1187-8. Montgomery F, S Willner. Screening for Idiopathic Scoliosis. Comparison of 90 cases shows less surgery by early diagnosis. Acta Orthop Scand 64(4) (1993) 456-458. Yawn BP, RA Yawn, D Hodge, et al. A population-based study of school scoliosis screening. JAMA 282 (1999) 1427–1432. Morais T, M Bernier, F Turcotte. Age- and Sex-specific Prevalence of Scoliosis and the Value of School Screening Programs. AJPH 75(12) (1985) 1377-80. Bunnel WP, Selective screening for scoliosis. Clin Orthop Relat Res 434 (2005) 40-5. Grivas TB, MH Wade, S Negrini, JP O'Brien, T Maruyama, M Rigo, HR Weiss, T Kotwicki, ES Vasiliadis, LS Neuhaus, T Neuhous, School Screening for Scoliosis. Where are we today? Proposal for a consensus. Scoliosis 2(1) (2007) 17. Burwell RG, The British Decision and Subsequent Events. Spine 13(10) (1988) 1192-94. Morrissy RT, School screening for scoliosis: A statement of the problem. Spine 13 (1988) 1195–1197. US Preventive Service Task Force: Screening for Idiopathic Scoliosis in Adolescent. Recommendation statement. Agency for Healthcare Research and Quality, Rockville, MD, 2004. Grivas TB, P Stavlas, K Koukos, P Samelis, B Polyzois, Scoliosis and Cavus Foot. Is there a Relationship? Study in Referrals, with and without Scoliosis, from School Screening. Stud Health Technol Inform 88 (2002) 10-14. Grivas TB, Samelis P, AS Pappa, P Stavlas, D Polyzois, Menarche in Scoliotic and Nonscoliotic Mediterranean Girls. Is There Any Relation between Menarche and Laterality of Scoliotic Curves? Stud Health Technol Inform 88 (2002) 30-6. Grivas TB, S Daggas, B Polyzois, P Samelis, The double rib contour sign in lateral spinal radiographs. Aetiologic implications for scoliosis? Stud Health Technol Inform 88 (2002) 38-43. Grivas TB, P Samelis, T Chatziargiropoulos, D Polyzois, Study of the rib cage deformity in children with 10º-20º of Cobb angle late onset idiopathic scoliosis, using rib vertebra angles. Stud Health Technol Inform 91 (2002) 20-24. Grivas TB, S Daggas, P Samelis, C Maziotou, P Kandris, Lateral spinal profile in school-screening referrals with and without late onset idiopathic scoliosis 10˚-20˚. Stud Health Technol Inform 91 (2002) 25-31. Grivas TB, A Arvaniti, C Maziotou, M Manesioti, A Fergadi, Comparison of body weight and height between normal and scoliotic children. Stud Health Technol Inform 91 (2002) 47-53. Dickson RA, SL Weinstein, Bracing (and Screening) – Yes or No? J Bone Joint Surg 81B(2) (1999) 193-98. Grivas TB, ES Vasiliadis, C Maziotou, OD Savvidou, The direct cost of Thriasio school screening program. Scoliosis 2(1) (2007) 7. Grivas TB, P Samelis, B Polyzois, B Giourelis, D Polyzois, School screening in the heavily industrialized area. Is there any role of industrial environmental factors in IS prevalence? Stud Health Technol Inform 91 (2002) 76-80. Grivas TB, E Vasiliadis, O Savvidou, M Mouzakis, G Koufopoulos, Geographic latitude and prevalence of adolescent idiopathic scoliosis. Stud Health Technol Inform 123 (2006) 84-89. Grivas TB, E Vasiliadis, V Mouzakis, C Mihas, G Koufopoulos, Association between adolescent idiopathic scoliosis prevalence and age at menarche in different geographic latitudes. Scoliosis 1(1) (2006) 9. Grivas TB, OD Savvidou, E Vasiliadis, S Psarakis, G Koufopoulos, Prevalence of scoliosis in women with Visual Deficiency. Stud Health Technol Inform 123 (2006) 52-56. Grivas TB, OD Savvidou, Melatonin the "light of night" in human biology and adolescent idiopathic. Scoliosis 2(1) (2007) 6. Grivas TB, ES Vasiliadis, VD Polyzois, V Mouzakis, Trunk asymmetry and handedness in 8245 school children. Pediatr Rehabil 9(3) (2006) 259-66.

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[24] Grivas TB, K Mihas, E Vasiliadis, C Maziotou, S Karathanou, V Polyzois, Handedness and laterality of trunk rotation in children screened at school for Scoliosis. J Bone Joint Surg 86-B Supp II (2004) 172. [25] Grivas TB, ES Vasiliadis, K Mihas, OD Savvidou, The effect of growth on the correlation between the spinal and rib cage deformity. Implications on idiopathic scoliosis pathogenesis. Scoliosis 2(1) (2007) 11. [26] Grivas TB, ES Vasiliadis, M Malakasis, M Mouzakis, D Segos, Intervertebral disc biomechanics in the pathogenesis of idiopathic scoliosis. Stud Health Technol Inform 123 (2006) 80-83.

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Suggestions for improvement of School Screening for Idiopathic Scoliosis T B. GRIVAS1, E S. VASILIADIS1, J P. O’BRIEN2 Scoliosis Clinic, Orthopaedic Department, “Thriasio” General Hospital, G. Genimata Avenue, Magula, 19600, Athens, Greece. 2 National Scoliosis Foundation (NSF), Boston, USA

1

Abstract. There is skepticism and the worth of school screening for the purposes of health care has been challenged. Numerous reasons are raised by the negativists to abandon these programs, even though the value of school screening is well documented in the literature. The aim of the present study is to update the evidence based recommendations for the improvement of school screening effectiveness, in order to support its continuation. All the relative research papers which originated from our scoliosis school screening program were analyzed. Specific suggestions for a) the organization, b) the optimal age of screening according to the geographical latitude, c) the best examined position, d) the standardization of referrals, e) the follow up of younger referrals with trunk asymmetry and f) the reduction of the financial cost are made. Today there is evidence that the incidence of surgery can significantly be reduced in areas where idiopathic scoliosis can be detected at an early stage through these programs. The introduction of these recommendations to all the existing school screening programs is strongly suggested, to reduce the negative impact they may have on families and on the health system and to improve their effectiveness. Keywords. Idiopathic scoliosis, school screening, improvement of school screening

1.

Introduction

There are numerous problems that prevent school screening for IS from being universally accepted. The low prevalence of IS, the high false positive referrals and the excessive cost, both direct and indirect, are raised by the negativists as reasons to abandon school screening programs. To our knowledge, there is no study in the literature to provide suggestions on how to improve the weak points of scoliosis school screening. The American Academy of Orthopaedic Surgeons, Scoliosis Research Society, Pediatric Orthopaedic Society of North America and American Academy of Pediatrics in their most recently published joint information statement on scoliosis screening do not support any recommendation against scoliosis screening, given the available literature [1]. Scoliosis screening has proven effective in many ways, and it is considered beneficial among the Orthopaedic community [2]. Furthermore, it provides the opportunity for early diagnosis and conservative treatment, which is often missed in the absence of screening [3]. The initial goal of scoliosis school screening was to detect IS at an early stage when deformity is likely to go unnoticed [3]. In an effort to enhance the effectiveness of school screening programs, the International Society on Scoliosis Orthopaedic and Rehabilitation Treatment (SOSORT) recommended as a new goal to identify those

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children who will be at risk for developing scoliosis [2] and those children who have curves likely to require conservative treatment [4]. This would require better organization, selective screening of high-risk children, improved examination techniques, standardization of the referral process and cost reduction. 2.

Material-Method

We reviewed all the research papers which originated from the “Thriasio” school screening program [5] and provided evidence based recommendations for the improvement of school screening effectiveness. 3.

Results

School screening has to be set up on a district basis after obtaining permission from the state authorities to approach and examine the school children. A permanent team of examiners should be created, after adequate experience. The examiners should have a core of 2-3 health professionals who are experienced in screening techniques, data entry and occasionally can be staffed by other health professionals on a voluntary basis. The examiners and all the involved individuals (children and their families) must have easy access to the referral hospital. It is important to inform and train if necessary all the interested parties in advance, by distribution of informative material and lectures. Before visiting a school the parents or carerers must fill a consent form (Europe) or be notified by letter as a courtesy in countries where school screening is legislated (North America) and the pupils must fill a particular form regarding their personal and demographic data. The facilities for examination should be prepared by the school stuff in advance and the timetable of that day should be modified accordingly, so that the examination time is kept effectively to a minimum. In a previous study we reported that the regression curve of both the IS prevalence and age at menarche by geographical latitude is following a parallel ascending course, especially in latitudes northern than 25o, which means that late age at menarche was parallel with higher prevalence of IS [6]. In order to increase the predictive value of school screening, we should screen children for scoliosis at a range of age adjusted for the geographical latitude. In a study of trunk asymmetry of 2071 children and adolescents we found a significant reduction in the proportion of asymmetry between the standing and sitting forward bending position during examination [7, 8]. By screening children in sitting position with the use of a scoliometer, the number of referrals can be decreased dramatically because the effect of leg length inequality and pelvic obliquity on the spine is eliminated. [8] Children with leg length inequality could be referred independently for further consultation. Sitting position reveals the true trunk asymmetry which could be associated with IS and therefore it is recommended as a standard examination method in a school screening program as well as a second level test for all potential referrals. It is essential when we set up the screening process to collect information about the gender, the chronological age, the age at menarche, the pattern and the magnitude of asymmetry and the growth potential. All these are very important prognostic factors for progression of a detected asymmetry. [9,10]. Trunk asymmetry should be measured by the use of a scoliometer in three sites of the spine (thoracic,

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thoracolumbar and lumbar) [7] and a cut-off point of 7o together with the prognostic factors should be used for referral. The same factors can be used in scoliosis clinic to determine whether it is necessary to x-ray a child or not, by the more experienced Orthopaedic surgeons, as a second stage of screening. In the model we propose [5] we standardize the referral process, by documenting all these factors, Table 1. Table 1. Recommended criteria for referral in scoliosis school screening 1. Trunk asymmetry (scoliometer reading) 5. Pattern of asymmetry

2. Gender 6. Growth potential

3. Chronological age 7. Maturation

4. Age at menarche for girls

Although asymmetry in skeletally mature children is unlikely to be associated with a progressive scoliotic curve, we noticed in a previous study that in some younger referred girls from the school-screening program there is a discrepancy between the thoracic scoliometer readings and the morphology of their spine [11]. After statistical analysis no significant correlation was found between the surface and the spinal deformity in the younger referred girls aged 8-13 years old, while in older referred girls aged 14-18 years old, this correlation was statistically significant. Therefore, all the younger individuals who are identified with a surface deformity but without a severe scoliotic curve are at risk for IS development and need to be kept under observation and not be discharged from regular follow up. The financial cost of a school screening program can be either direct or indirect [12]. There is no general consensus among economists as to what constitutes the indirect cost in a cost effectiveness analysis because the indirect cost cannot be measured accurately as it is related to the effectiveness of the school screening program. A more effective screening program has lower indirect cost. Therefore the economic information on screening for scoliosis which is available to decision-makers should mainly be based on studies of the direct cost of such programs. In a six year performance of the “Thriasio” school screening program, the direct cost for the examination of each child was 2.04 €, which is considered low [5]. This study provides evidence that the direct cost of a screening program can be reduced to a minimum, if it is well organized and it is performed according to the model we propose [5]. 4.

Discussion

School screening programs are performed in different ways around the world. Unfortunately there is no consensus among the experts on specific criteria of screening. As a consequence of the luck of standards of scoliosis screening, there is no adequate evidence based on outcome results necessary to either enhance or eliminate the school screening process. Today there is evidence that the incidence of surgery can significantly be reduced where conservative treatment is available on a high standard [13, 14, 15]. In areas where scoliosis school screening has stopped, the referral mechanisms for IS are leading to a suboptimal case-mix in orthopaedics in terms of appropriateness of referral and resulted in increased late referrals with regards to brace treatment indications [16]. School screening through detection of IS at an early stage is the only tool we have to detect mild and moderate spinal curves which can be treated conservatively in an effective way. Physicians should be more interested in quality of care for their patients

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than in epidemiology, numbers, statistics, or money. Prevention instead of treatment, which was an axiom in Ancient Greece, must return as a standard health policy in civilized societies. And school screening is primarily a preventive process. Our screening vision should be strongly focused on how to improve its effectiveness so that we can change the paradigm from today’s “correction” and “fusion” goal to a stronger goal of maintaining the natural shape and mobility of the spine and “preventing” a spinal deformity from developing. References [1]

[2]

[3] [4] [5] [6]

[7] [8] [9] [10] [11]

[12] [13] [14] [15]

[16]

Information Statement: Screening for idiopathic scoliosis in adolescents. American Academy of Orthopaedic Surgeons (AAOS), Scoliosis Research Society (SRS), Pediatric Orthopaedic Society of North America (POSNA) and American Academy of Pediatrics (AAP), October 1, 2007. Grivas TB, MH Wade, S Negrini, JP O'Brien, T Maruyama, M Rigo, HR Weiss, T Kotwicki, ES Vasiliadis, LS Neuhaus, T Neuhous, School Screening for Scoliosis. Where are we today? Proposal for a consensus. Scoliosis 2(1) (2007) 17. Bunnel WP, Selective screening for scoliosis. Clin Orthop Relat Res 434 (2005) 40-5. Weiss HR, S Negrini, M Rigo, T Kotwicki, MC Hawes, TB Grivas, T Maruyama, F Landauer, Indications for conservative management of scoliosis (guidelines). Scoliosis 1(1) (2006) 5. Grivas TB, ES Vasiliadis, C Maziotou, OD Savvidou, The direct cost of “Thriasio” school screening program. Scoliosis 2(1) (2007) 7. Grivas TB, E Vasiliadis, V Mouzakis, C Mihas, G Koufopoulos, Association between adolescent idiopathic scoliosis prevalence and age at menarche in different geographic latitudes. Scoliosis 1(1) (2006) 9. Grivas TB, ES Vasiliadis, VD Polyzois, V Mouzakis, Trunk asymmetry and handedness in 8245 school children. Pediatr Rehabil 9(3) (2006) 259-66. Grivas TB, E Vasiliadis, G Koufopoulos, D. Segos, G Triantafilopoulos, V Mouzakis, Study of trunk asymmetry in normal children and adolescents. Scoliosis 1(1) (2006) 19. Bunnell WP, The natural history of idiopathic scoliosis before skeletal maturity. Spine 11 (1986) 773– 776. Soucacos PN, K Zacharis, K Soultanis, J Gelalis, Xenakis T, Beris AE, Risk factors for idiopathic scoliosis: Review of a 6-year prospective study. Orthopedics 23 (2000) 833–838. Grivas TB, ES Vasiliadis, K Mihas, OD Savvidou, The effect of growth on the correlation between the spinal and rib cage deformity. Implications on idiopathic scoliosis pathogenesis. Scoliosis 2(1) (2007) 11. Montgomery F, U Persson, G Benoni, S Willner, B Lindgern, Screening for scoliosis. A costeffectiveness analysis. Spine (1990) 15(2) 67-70. Weiss WR, G Weiss, HJ Schaar, Incidence of surgery in conservatively treated patients with scoliosis. Pediatr Rehabil 6(2) (2003) 111-8. Rigo M, C Reiter, HR, Effect of conservative management on the prevalence of surgery in patients with adolescent idiopathic scoliosis. Pediatr Rehabil 6(3-4) (2003) 209-14. Maruyama T, T Kitagawa, K Takeshita, K Mochizuki, K Nakamura, Conservative treatment for adolescent idiopathic scoliosis: can it reduce the incidence of surgical treatment? Pediatr Rehabil 6(34) (2003) 215-9. Beausejour M, M Roy-Beaudry, L Goulet, H Labelle, Patient characteristics at the initial visit to a scoliosis clinic: a cross-sectional study in a community without school screening. Spine 32 (2007) 1349-1354.

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A Machine Learning Approach to Assess Changes in Scoliosis L RAMIREZ 1, N G. DURDLE 1, V. J RASO 2 1

Department of Electrical and Computer Engineering, University of Alberta, Canada 2 Glenrose Rehabilitation Hospital, Capital Health Authority, Canada

Abstract. This paper presents a machine learning approach that can be used to evaluate the validity of the results obtained with an automated system to measure changes in scoliotic curves. The automated system was used to measure the inclinations of 141 vertebral endplates in spine radiographs of patients with scoliosis. The resulting dataset was divided into training and test set. The training set was used to configure three classifiers: a support vector classifier (SVC), a decision tree classifier (DT) and a logistic regression classifier (LR). Their performance was evaluated on the test set. The SVC had an accuracy of 86% discriminating Good Results (those in which the error was less than 3°) from Bad Results. This accuracy was better than that of the LR (76%) and DT (68%). The differentiation between Good and Bad Results using the proposed machine learning approach was achieved successfully. Keywords. Scoliosis, image registration, machine learning

1. Introduction Scoliosis is a condition that involves a lateral curvature and rotation of the spine that could cause noticeable trunk deformities. This condition affects between 2% and 4% of adolescents [1] and between 70% and 80% of the cases have an unknown cause [2]. The management of patients with scoliosis relies on subjective identification and measurement of a set of features on spine radiographs. These include: the Cobb angle [3], vertebral endplate tilt angles, and the apical and end vertebrae of the scoliotic curve. Because these features are assessed manually, they are prone to high inter- and intraobserver variability [4]. For instance, the typical inter- and intra-observer variability of the Cobb angle is accepted to be +/- 5 degrees, which is comparable to the threshold of change that is considered when making treatment decisions. In this paper, a machine learning approach that can be used to automatically assess the quality of the results obtained with an automated system to measure changes in scoliotic curves is presented. Since the clinical focus was on spinal deformities, the main interest was on measuring the inclinations of vertebrae in spine radiographs. Machine learning is concerned with the design and development of techniques that allow computers to learn from datasets. Among machine learning techniques, classification strategies can be used to discriminate between groups. In this work, a classification strategies is proposed to discriminate Good Results (those in which the inclination measurement had an error greater than 3 degrees) from Bad Results. The proposed classification strategy is based on Support Vector Classifiers (SVC) [5]. The

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main goal was to determine whether SVC could predict results quality sufficiently well to be used in clinical practice. SVC were chosen because they usually outperform techniques such as Artificial Neural Networks when trained using small datasets as is usually the case in scoliosis research. Moreover, unpublished preliminary studies comparing the performance of radial basis function neural networks [6] and SVC indicated the superiority of the latter in our datasets of scoliosis patients. Finally, the results of applying a SVC to the dataset of scoliosis patients are compared to those obtained by applying logistic regression [7] and classification decision trees [8] to the dataset. 2. Methods Retrospectively, radiographs of patients with scoliosis from the database of the scoliosis clinic at the Glenrose Rehabilitation Hospital (Edmonton, Canada) were examined, after getting Ethics Approval, to select patients for the study. The following inclusion criteria were used: 1) having available a posterior-anterior standing radiograph with a maximum Cobb angle of less than 75°; 2) not having undergone surgery; and 3) having at least a visually identifiable vertebral endplate. Eighteen patients and a total of 141 vertebral endplates satisfied the inclusion criteria. Patients had a variety of spinal curvatures, with an average of maximum Cobb angle of 39° ± 18° (range 8°-74°). There were 7 (39%) patients with double curve and 11 (61%) patients with single curves. All images were carefully annotated to create the “ground truth”. A graphical user interface was used to allow the user to select two vertebral endplates to be analyzed (Figure 1). Based on the user selection, two regions of interest (ROIs) were created (Figures 2 and 3). A model (Figure 4) was then registered to the ROIs, using custom-made software, to find the location and orientation of the endplates under study (See Figure 5 for two unsuccessful registrations and Figure 6 for two successful registrations). The process was repeated for an average of 8 vertebrae per radiograph creating a database of 141 registration solutions. The data set for the experiments was divided into training and test set according to the time in which the data became available. The training set consisted of 14 radiographs from which 104 vertebral endplates were selected using the selection criteria previously discussed. The test set consisted of 4 radiographs that became available after the first group. From the second group of radiographs, 37 vertebral endplates were selected following the inclusion criteria previously discussed. Once the endplates were selected, model-to-image registrations were performed. After registration, each solution was evaluated with respect to the ground truth. If the difference in the rotation angle was greater than 3° the solution was labeled as a Bad Solution. Otherwise, the location of the registered model was assessed visually to assign one of two possible labels: Bad Results (BR) or Good Results (GR). For the training set there were 54 registration solutions labeled as BR and 50 registration solutions labeled as GR. For test set there were 25 registration solutions labeled as BR and 12 registration solutions labeled as GR. The training set was used to build three classifiers: a Support Vector Classifier (SVC), a Decision Tree Classifier (DT), and a Logistic Regression Classifier (LR). The performance of the classifiers was assessed using the test set.

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Figure 1. Spine radiograph with user-entered landmarks (indicated with a *)

Figure 2. Region of interest around top landmark

Figure 3. Region of interest around bottom landmark

Figure 4. Model used for registration

L. Ramirez et al. / A Machine Learning Approach to Assess Changes in Scoliosis

Figure 5. Two un-successful registrations (indicated with straight lines)

Figure 6. Two successful registrations (indicated with straight lines)

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3. Results This section compares the results, in testing, of the Support Vector Classifier (SVC) with those of the Decision Tree (DT) and Logistic Regression (LR) classifiers and evaluates their performance. The results of classifying the cases into Good Results (GR) and Bad Results (BR) (using the test data set) are summarized in Table 1. In terms of accuracy, the SVC outperformed the DT and LR classifiers with accuracy of 86% compared to 68% and 76%, respectively, for the DT and LR. Moreover, the SVC detected the largest percentage of cases with BR: 96%, compared to 64% of the LR and only 52% of the DT. Table 1. Classification problem: Bad Results versus Good Results. Test results with the SVC, DT, and LR classifiers on the test set. TO: True Output. CO: Classifier Output. BR: Bad Results. GR: Good Results.

TO\CO BR GR Accuracy

BR 24(96%) 4(33%)

SVC GR 1(4%) 8(67%) 86%

DT BR 13(52%) 0(0%)

GR 12(48%) 12(100%) 68%

LR BR 16(64%) 0(0%)

GR 9(36%) 12(100%) 76%

4. Discussion Results obtained from comparing classifiers with a test set showed that the support vector outperformed a decision tree and logistic regression classifiers. The classification accuracy for the SVC was 86% in testing. The classification accuracy for the DT was 68% in testing. The classification accuracy for the LR was 76% in testing. Even though, the SVC achieved a high discriminatory power to Bad Results, work on improving its discriminatory power continues because improvements in it will translate to early detection of Bad Results guaranteeing a high level of care for the patients. It was observed that 1 record in the test set was misclassified by all the classifiers. This may suggest that the misclassified record was an outlier present in the data set. Although the SVC-based method was designed for estimating the quality of model-to-image registrations of spine radiographs, the proposed approach may be useful in other contexts as well. 5. Conclusion In conclusion, it is possible to differentiate Good Results from Bad Results by using the proposed support vector classifier. This finding may be useful in identifying when an application for automatically measuring scoliosis severity requires domain expert intervention to provide new configuration settings that could improve the results 6. Acknowledgement This work benefited from economic support from the Alberta Provincial CIHR Training Program in Bone and Joint Health, NSERC, and Edmonton Orthopaedics Research Committee.

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References [1] [2] [3] [4] [5] [6] [7]

[8]

Roach J.W., Adolescent idiopathic scoliosis, Orthop. Clin. N. Am., 1999; 30: 353-365. Khouri N., et al., Idiopathic scoliosis. Strategy, pathophysiology, and deformity analysis, EMC. Rhumatologie Orthopédie, 2004; 1: 17-44. Cobb J. R., Outline for the study of scoliosis, instructional course lectures, The American Academy of Orthopedic Surgeons, 1948; 5: 261-275. Morrissy R. T., et al. Measurement of the Cobb angle on radiographs of patients who have scoliosis. Evaluation of intrinsic error, J. Bone Joint Surg. Am, 1983; 65: 1302-1313. Vapnik V.N., “An overview of statistical learning theory,” IEEE Transactions on Neural Networks, vol. 10, no. 5, pp. 988-999, 1999. Bishop C.M., Neural Networks for Pattern Recognition. Oxford, U.K.: Claredon, 1995. Smith A.E., Nugent C. D., McClean S. I., “Evaluation of inherent performance of intelligent medical decision support systems: Utilizing neural networks as an example,” Artificial Intelligence in Medicine, vol. 27, no. 1, pp. 1–27, 2003. Cios K., Pedrycz W., Swiniarski R., Data Mining Methods for Knowledge Discovery. Boston: Kluwer Academic Publishers , 1998.

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Chapter 6 Mathematical Modelling

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Chapter 8 Treatment of Scoliosis

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Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved. doi:10.3233/978-1-58603-888-5-289

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Thoracoplasty in the Surgical Treatment of Adolescent Idiopathic Scoliosis T. GREGGI, G. BAKALOUDIS, F. LOLLI, M. DI SILVESTRE, A. CIONI, S. GIACOMINI, G. BARBANTI BRODANO, F. VOMMARO, K. MARTIKOS, P. PARISINi Spine Surgery Department, Istituto Ortopedico Rizzoli, Bologna ,Italy Abstract. A consecutive series of 40 adolescents surgically treated between 1998-2001, by posterior spinal fusion and thoracoplasty were compared with a similar group of 40 adolescents treated in the same period by posterior only segmental fusion. Clinical and radiographic analysis was performed, including the SRS-30 questionnaire and Pulmonary Function Tests (PFT). Minimum five years follow-up was requested. No statistical differences were found between the two groups in PFT’s both pre-operatively and at latest follow up. Our findings suggest that thoracoplasty did not adversely affected long-term PFT’s in AIS patients treated by posterior spinal fusion alone.

1.

Introduction

The primary goal of surgical treatment in adolescent idiopathic scoliosis (AIS) remains the arrest of further curve progression by obtaining a solid arthrodesis mass, long-term balance of the spine in both the sagittal and coronal planes, and preservation of the maximum number of motion segments. Several additional factors, e.g., safety, cost, and morbidity, are important in the surgical decision-making. In recent years, with the advent of modern third-generation segmental posterior instrumentation, in particular pedicle screws only constructs, a significant deformity correction is also desired by both the physician and the patient, whereas preservation or enhancement of pulmonary function is a strong consideration for thoracic curves [1-5]. The association between a spinal deformity and pulmonary impairment has been widely reported [6-13], with more severe curves (>100°) considered to be responsible for a significant decrease in clinical pulmonary function. A recent multicenter study on 631 AIS patients found a moderate or severe pulmonary impairment in association with thoracic curves of <50°, showing that some patients with adolescent idiopathic scoliosis may have clinically relevant pulmonary impairment that is out of proportion with the severity of the spinal deformity [14]. Posterior spinal arthrodesis with thoracoplasty and an open anterior approach, with respect to a posterior only fusion, were found to have a deleterious effect on pulmonary function for as long as five years postoperatively and chest cage preservation was recommended to maximize both absolute and percent-predicted pulmonary function values after surgical treatment of adolescent idiopathic scoliosis [15]. More recently, both anterior open approaches and thoracoplasty were the only variables found to be related to a clinically significant reduction in the predicted 2-year pulmonary function in surgically treated AIS patients. The magnitude of the effects of both these variables, however, was considered modest [16].

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The aim of the present study was to compare two similar groups of adolescents surgically treated for their spinal deformity, the first by means of posterior only segmental fusion (PSF), the second by posterior spinal fusion and thoracoplasty (PSF+T), focusing on the long-term effects of thoracoplasty in the surgical treatment of adolescent idiopathic scoliosis. 2.

Materials and Method

A consecutive series of 40 patients with main thoracic AIS curves (Lenke type 1, 2, 3, and 4), surgically treated between 1998 - 2001 at one institution by PSF+T were compared with a similar group of 40 adolescents treated in the same period by PSF alone. Inpatient and outpatient charts were used for the collection of demographic data, and annotation of any medical and surgical-related complications, including revision surgeries. The Lenke surgical classification of AIS [17] was used to describe curve patterns. Radiographic evaluation on standing postero-anterior and lateral films on long-cassette (90x30 cm) was performed, including Cobb measurements [18] of the major thoracic (MT) curve, and thoracic kyphosis (T5–T12), both preoperatively and postoperatively, and at the latest follow up visit. A radiographic Rib Hump (RH) assessment (Fig.1), as proposed by Potter [19], was also performed. On the final visit, the SRS-30 questionnaire was administered, and a Pulmonary Function Test (PFT) evaluation was performed. A minimum five years’ follow-up was requested for inclusion in the study. During the period considered for the current investigation the same surgical team (three different surgeons) performed all surgeries, with indication for thoracoplasty being a clinically relevant rib hump (i.e > 15° on the scoliometer measurement), or a particular concern of such deformity from the patient or family. Statistical analysis was performed using the t-test (paired and unpaired), the Wilcoxon test for non-parametric paired analysis, and the Mann-Whitney test for nonparametric unpaired analysis. Results are expressed as the mean (SD), with a P value < 0.05 considered as being statistically significant.

Fig. 1 Diagram showing the measurement technique for assessing the RH deformity. The RH is the linear distance between the left and right posterior rib prominences at the apex of the rib deformity on a lateral radiograph.

T. Greggi et al. / Thoracoplasty in the Surgical Treatment of Adolescent Idiopathic Scoliosis

3.

291

Results

We were able to review the entire series at an average clinical follow of 6.2 years (1.3). There were no statistically significant differences between the two groups concerning gender, age (PSF+T:16.3 years vs PSF: 15.2 years), Lenke’s curve type distribution, and Cobb’s preoperative main thoracic (MT) curve magnitude (PSF+T: 66° vs PSF: 63°), whereas both final MT percent correction (PSF+T: 53.03% vs PSF: 51.35%; p<0.03) and RH absolute correction (PSF+T: -2.1 cm vs PSF: -1.5; p<0.01) were superior in the thoracoplasty group (Table 1). We found no statistically significant differences between the two groups in terms of sagittal contour, with thoracic kyphosis (T5–T12) angle similar in each group before surgery (PSF+T 35.32° vs PSF 35.22°), immediate postoperative (26° vs 29.3°), and at final follow-up evaluation (28.4° vs 32.4°). Table 1 Gender (M/F) Age (years) Lenke’s distribution Preoperative main thoracic Cobb Follow-up main thoracic Cobb Percent overall correction Preoperative kyphosis (T5-T12) Follow-up kyphosis (T5-T12) Preoperative Rib hump (cm) Postoperative Rib hump (cm) Overall Rib hump correction (cm)

PSF+T group 6M/34F 16.3 (2) 25 “1”/10 “2”/5 “3” 66 (14) 31 (11.2) 53.03 (12.6) 35.32 (10.1) 28.4 (9.8) 3.45 (0.44) 1.69 (0.77) -2.1 (0.9)

PSF group 5M/35F 15.2 (1.9) 29 “1”/8 “2”/3 “3” 63 (13) 35 (13) 51.35 (12.8) 35.22 (12.5) 32.4 (10.6) 2.96 (0.63) 1.91 (0.4) - 1.5 (1.1)

P n.s n.s n.s n.s n.s n.s n.s n.s n.s n.s < 0.01

With regards to PFT’s, in the PSF+T group both the absolute values of forced vital capacity was unchanged, from 2.87 to 3.1 L (p = 0.16), and those of forced expiratory volume in one second from 2.39 to 2.60 L (p = 0.36), in the pre-operative and last followup evaluation. In the PSF group the forced vital capacity increased from 2.83 to 3.34 L [p < 0.0001] and forced expiratory volume in one second increased from 2.41 to 2.84 L [p < 0.0001]), showing nevertheless no statistical differences both pre-operatively and at latest follow up when the two groups were compared (Table 2). At the latest follow-up visit, SRS-30 scores did not show any statistical differences between the two groups (total score PSF+T: 4.1 vs PSF:4.3; n.s) (Table 2). Table 2 Preoperative FVC (L)/ FVC % Preoperative FEV1 (L)/ FEV1 % Follow-up FVC (L)/ FVC % Follow-up FEV1 (L)/ FEV1 % Follow-up vs Pre-operative FVC (L)/ FVC % Follow-up vs Pre-operative FEV1 (L)/ FEV1 % SRS pain SRS self-image SRS function SRS mental health SRS satisfaction SRS total score

PSF+T group 2.87 (0.75) / 85% (15) 2.39 (0.48) / 82% (12) 3.1 (0.6) / 83% (14) 2.60 (0.36) / 80% (11.5) 4.16 (0.65) 3.84 (0.53) 3.54 (0.71) 4.02 (0.35) 4.3 (0.75) 4.1 (0.25)

PSF group 2.83 (0.8) / 84% (14) 2.41 (0.7) / 80% (13) 3.34 (0.5) / 87% (18) 2.84 (0.4) / 83% (17) < 0.0001 < 0.0001 4.23 (0.54) 3.46 (0.71) 3.25 (0.36) 3.63 (0.46) 4.5 (0.29) 4.3 (0.30)

P n.s n.s n.s n.s n.a n.a n.s n.s n.s n.s n.s n.s

No fatal complications or neurologic injuries, either acute or delayed deep wound infections were observed in this case series. In the PSF+T group 3 surgery-related

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complications (proximal hook disengagement) in 3 patients (7.5%) required 2 revision surgeries in 2 patients (at 3 ½, and 5 years respectively from the index procedure), whereas post-operative thoracoplasty-related pulmonary complications were encountered in 10 patients (moderate pleural effusion in 7, pneumonia secondary to moderate atelectasis in 2, pneumothorax in 1), requiring chest tube placement in 3 patients. In the PSF group a revision surgery with complete removal of the instrumentation was performed in a 19-year-old female patient due to persistent late operative site pain; intra-operative cultures were negative, 4 years after the index procedure. Three post-operative medical complications were observed in this latter group (1 acute cholecistitis, a superior mesenteric artery syndrome, a moderate pneumonia in 1) that did not require any invasive procedure and healed uneventfully by conservative treatment. 4.

Conclusions

According to our findings thoracoplasty did not adversely affect long-term PFT’s in AIS patients treated by posterior spinal fusion alone, as suggested by previous reports. A relatively high incidence of peri-operative complications were observed in the thoracoplasty group when compared to the posterior only fusion group. A trend towards better coronal plain correction and rib hump amelioration was found, not clearly reported by a self-assessment disease specific questionnaire. References [1] [2] [3] [4] [5] [6] [7] [8] [9]

[10] [11] [12] [13] [14] [15]

Bridwell K Surgical Treatment of Idiopathic Adolescent Scoliosis. Spine 24 (1999) 2607-2616. Lenke LG Betz R.et al., Spontaneous Lumbar Curve Coronal correction After Selective Anterior or Posterior Thoracic Fusion in Adolescent Idiopathic Scoliosis. Spine 24 (1999) 1663-1672. Haher TR, Merola A, et al., Metaanalysis of surgical outcome in adolescent idiopathic scoliosis. A 35year English literature review of 11,000 patients. Spine 20 (1995) 1575–1584. Kim YJ, Lenke LG, et al., Comparative Analysis of Pedicle Screw Versus Hybrid Instrumentation in Posterior Spinal Fusion of Adolescent Idiopathic Scoliosis. Spine 31 (2006) 291-298. Suk SI, Lee CK, et al., Segmental pedicle screw fixation in the treatment of thoracic idiopathic scoliosis. Spine 20 (1995) 1399-1405. Aaro S, Ohlund C, Scoliosis and pulmonary function. Spine 9 (1984) 220–222. Leong JC, Lu WW, et al., Kinematics of the chest cage and spine during breathing in healthy individuals and in patients with adolescent idiopathic scoliosis. Spine 24 (1999) 1310–1315. Vedantam R, Crawford AH, The role of preoperative pulmonary function tests in patients with adolescent idiopathic scoliosis undergoing posterior spinal fusion. Spine 22 (1997) 2731–2734. Vedantam R, Lenke LG, et al., A prospective evaluation of pulmonary function in patients with adolescent idiopathic scoliosis relative to the surgical approach used for spinal arthrodesis. Spine 25 (2000) 82–90. Wood KB, Schendel MJ, et al., Thoracic volume changes in scoliosis surgery. Spine 21 (1996) 718–723. Weinstein SL, Zavala DC, Ponseti IV, Idiopathic scoliosis: long-term follow-up and prognosis in untreated patients. J Bone Joint Surg Am 63 (1981) 702–712. Upadhyay SS, Mullaji AB, et al., Relation of spinal and thoracic cage deformities and their flexibilities with altered pulmonary functions in adolescent idiopathic scoliosis. Spine 20 (1995) 2415–2420. Kearon C, Viviani GR, et al., Factors determining pulmonary function in adolescent idiopathic thoracic scoliosis. Am Rev Respir Dis 148 (1993) 288–294. Newton PO, Faro FD et al., Results of Preoperative Pulmonary Function Testing of Adolescents with Idiopathic Scoliosis. A Study of Six Hundred and Thirty-one Patients. J Bone Joint Surg [Am] 87 (2005) 1937-1946. Kim YJ Lenke LG et al., Prospective evaluation of pulmonary function in adolescent idiopathic scoliosis relative to surgical approach: minimum 5-year follow-up. J Bone Joint Surg [Am] 87 (2005) 1534-1541.

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Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved. doi:10.3233/978-1-58603-888-5-310

Congenital scoliosis – presentation of three severe cases treated conservatively H-R WEISS Asklepios Katharina Schroth Spinal Deformities Rehabilitation Centre, Korczakstr. 2, 55566 Bad Sobernheim, Germany, [email protected]

Abstract. In view of the very limited data about conservative treatment of patients with congenital scoliosis (CS) available, early surgery is suggested already in mild cases with formation failures in the first three years of life. It is common sense that patients with failures of segmentation will not benefit from conservative treatment at all and the same applies to failures of formation with curves of > 50 degrees in infancy. Materials and Methods. Two patients with rib synostosis denied surgery before entering the pubertal growth spurt. These patients have been treated conservatively with braces and Scoliosis In-Patient Rehabilitation (SIR) and now are beyond the pubertal growth spurt. One patient with a formation failure and a curve of > 50 degrees lumbar has been treated with the help of braces and physiotherapy from 1.6 years on and is still under treatment now at the age of 15 years. Results. Severe decompensation was prevented in the two patients with failure of segmentation, however a severe thoracic deformity is evident with underdeveloped lung function and severe restrictive ventilation disorder. The patient with failure of formation is well developed, now without cosmetic or physical complaints although his curve progressed at the end of the growth spurt due to final mal-compliance. Conclusions. Failures of segmentation should be advised to have surgery before entering the pubertal growth spurt. In case they deny, conservative treatment can at least in part be beneficial. For patients with failures of formation conservative treatment should be suggested in the first place because long-term outcomes of early surgery beyond pubertal growth spurt are not yet revealed. Keywords. Congenital scoliosis, conservative treatment, indication for surgery

1.

Introduction

In view of the very limited data about conservative treatment of patients with congenital scoliosis (CS) available, early surgery is suggested already in mild cases with formation failures in the first three years of life. It is common sense that patients with failures of segmentation will not benefit from conservative treatment at all and the same applies to failures of formation with curves of > 50 degrees in infancy. Purpose of this study was to reveal the effects of conservative treatment in this rare patient population.

H.-R. Weiss / Congenital Scoliosis – Presentation of Three Severe Cases Treated Conservatively

2.

311

Material and Method

Two patients with rib synostosis (Fig. 1 and 2) denied surgery before entering the pubertal growth spurt. These patients have been treated conservatively with braces and Scoliosis InPatient Rehabilitation (SIR) and now are beyond the pubertal growth spurt. One patient with a formation failure (Fig. 3) and a curve of > 50 degrees lumbar has been treated with the help of braces and physiotherapy from 1.6 years on and is still under treatment now at the age of 15 years.

3.

Results

Severe decompensation was prevented in the two patients with failures of segmentation, however a severe thoracic deformity is evident with underdeveloped lung function and severe restrictive ventilation disorder. The patient with failure of formation is well developed now without cosmetic or physical complaints although his curve progressed at the end of the growth spurt due to mal-compliance.

Figure 1: Follow-up from 10 years to 18 years. Progression from 10–12 years, after that stable. At 10 years the patient had 62° and progressed to 71° at the age of 12. Last x-ray: 72°. VC: 650 ml / 19% of the predicted value.

4.

Discussion

So called long-term studies reporting on congenital scoliosis patients treated surgically reveal follow-up periods of 3-6 years with most of the patients being still before the pubertal growth spurt at final follow-up [1-4]. While the complications reported for the entity of congenital scoliosis varies widely between 0 and 48% in short- to mid-term [5-8]. The long-term complication rate for congenital scoliosis patients operated on is not yet reported.

312

H.-R. Weiss / Congenital Scoliosis – Presentation of Three Severe Cases Treated Conservatively

Figure 2: From 2002 (64°) before pubertal growth spurt until 2007 (59°) at Risser 4 (15 years.) no progression has been detected. The ATR has been reduced with the Chêneau braces applied from initially 17° to 9° in January 2008 during the last SIR.VC: 1.640 ml / 33% of the predicted value.

Figure 3: 52° at the age of 18 months, in between the curve without brace on was down to 40°, 58° at final follow-up with Risser 4 after final mal-compliance. The patient had no cosmetic complaints. A small lumbar hump is visible but no decompensation.

To conclude from single case reports that: “The early fusion prevented the customary severe progression of this condition and early death due to cor pulmonale ”, somehow seems biased pro surgery when there could be the possibility that even without surgery cor pulmonale would not necessarily be the consequence of an untreated congenital scoliosis [9,10]. On the other hand the patients reported on in these case reports are not yet beyond 50 years of age and might develop cor pulmonale in the future.

H.-R. Weiss / Congenital Scoliosis – Presentation of Three Severe Cases Treated Conservatively

5.

313

Conclusions

Failures of segmentation should be advised to have surgery before entering the pubertal growth spurt. In case they deny, conservative treatment can at least in part be beneficial. For patients with failures of formation conservative treatment should be suggested in the first place because long-term outcomes of early surgery beyond pubertal growth spurt in adulthood are not yet revealed.

References [1]

Thompson AG, Marks DS, Sayampanathan SR, Piggott H: Long-term results of combined anterior and posterior convex epiphysiodesis for congenital scoliosis due to hemivertebrae. Spine. 1995 Jun 15;20(12):1380-5. [2] Marks DS, Sayampanathan SR, Thompson AG, Piggott H: Long-term results of convex epiphysiodesis for congenital scoliosis. Eur Spine J. 1995;4(5):296-301. [3] Bollini G, Docquier PL, Viehweger E, Launay F, Jouve JL: Thoracolumbar hemivertebrae resection by double approach in a single procedure: long-term follow-up. Spine. 2006 Jul 1;31(15):1745-57. [4] Lazar RD, Hall JE: Simultaneous anterior and posterior hemivertebra excision. Clin Orthop Relat Res. 1999 Jul;(364):76-84. [5] Ayvaz M, Alanay A, Yazici M, Acaroglu E, Akalan N, Aksoy C. Safety and efficacy of posterior instrumentation for patients with congenital scoliosis and spinal dysraphism. J Pediatr Orthop. 2007 Jun;27(4):380-6. [6] Benli IT, Duman E, Akalin S, Kiş M, Aydin E, Un A. [An evaluation of the types and the results of surgical treatment for congenital scoliosis]. Acta Orthop Traumatol Turc. 2003;37(4):284-98. [7] Shono Y, Abumi K, Kaneda K. One-stage posterior hemivertebra resection and correction using segmental posterior instrumentation. Spine. 2001 Apr 1;26(7):752-7. [8] Kahanovitz N, Brown JC, Bonnett CA.The operative treatment of congenital scoliosis. A report of 23 patients. Clin Orthop Relat Res. 1979 Sep;(143):174-82. [9] Winter RB, Lonstein JE. Congenital scoliosis with posterior spinal arthrodesis T2-L3 at age 3 years with 41year follow-up. A case report. Spine. 1999 Jan 15;24(2):194-7. [10] Winter RB, Lonstein JE. Congenital thoracic scoliosis with unilateral unsegmented bar and concave fused ribs: rib osteotomy and posterior fusion at 1 year old, anterior and posterior fusion at 5 years old with a 36year follow-up. Spine. 2007 Dec 15;32(26):E841-4.

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Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved. doi:10.3233/978-1-58603-888-5-314

Conservative scoliosis treatment in patients with Prader-Willi syndrome H-R WEISS, S BOHR Asklepios Katharina Schroth Spinal Deformities Rehabilitation Centre, Korczakstr. 2, 55566 Bad Sobernheim, Germany, [email protected]

Abstract. Patients with Prader-Willi syndrome often suffer from scoliosis of major degrees. Due to current literature surgical intervention seems the gold standard of treatment although the rate of complications in this condition are reported to be significantly higher than in patients with Adolescent Idiopathic Scoliosis. Purpose of this study was to reveal the effects of conservative treatment in this rare patient population. Materials and Methods. A case series of patients with this condition has been investigated to estimate as to whether Prader-Willi patients with scoliosis may benefit from conservative scoliosis management. 9 Patients with this condition have been found in our out-patient database. 5 of these retarded patients (3 girls, two boys) today are 19 years and older and therefore are without any significant residual growth. Average Cobb angle was 47 degrees (34 – 66 degrees) at 12 years, average observation time was 6.4 years. Results. Two of the five patients progressed. Average Cobb angle after follow-up was 52 degrees. No progression beyond 70 degrees has been found after cessation of growth. In one patient the curve deteriorated clearly after reducing brace wearing time and therefore was due to non-compliance. Conclusions. Stabilisation of scoliosis due to Prader-Willi syndrome is possible by means of conservative management. To expose this patient population to the risks of surgical management seems not to be justified. Keywords. Prader-Willi syndrome, scoliosis, surgery, complications, indications

1.

Background

Children with Prader-Willi syndrome frequently have musculo-skeletal problems such as joint hyperlaxity, hypotonia, delayed bone age, and scoliosis. Their musculo-skeletal problems are magnified by the extreme obesity many of these patients exhibit [1]. Osteopenia, poor impulse control and defiant behaviors, and diminished pain sensitivity are aspects of PWS that may complicate all facets of orthopaedic nonsurgical and surgical management in this patient population. The treating orthopaedic surgeon must plan carefully and proceed with caution when treating children and adults with PWS [2].

315

H.-R. Weiss and S. Bohr / Conservative Scoliosis Treatment

Patients with Prader-Willi Syndrome often suffer from scoliosis of major degrees. Due to current literature surgical intervention [3] seems the gold standard of treatment although the rates of complications in this condition are reported to be significantly higher than in patients with Adolescent Idiopathic Scoliosis. Purpose of this study was to reveal the effects of conservative treatment in this rare patient population.

2.

Material and Methods

A case series of patients with this condition has been investigated to estimate as to whether Prader-Willi patients with scoliosis may benefit from conservative scoliosis management. 9 Patients with this condition have been found in our out-patient database. 5 of these retarded patients (3 girls, two boys) today are 19 years and older and therefore are without any significant residual growth. Average Cobb angle was 47 degrees (34 – 66 degrees), average observation time was 6.4 years.

19 / 17°

30 / 40°

i. Br. 17 / 25°

29 / 37°

24 / 30°

Figure 1. Left: First presentation at the age of 11 years, 2nd. from left: Brace indication at 12 years, middle: In brace correction, 2nd. from right: At weaning at the age of 20 years and on the right: Four years without brace at 24 years. During the last 4 years the woman lost weight.

3.

Results

Two of the five patients progressed. Average Cobb angle after follow-up was 52 degrees. No progression beyond 70 degrees has been found after cessation of growth. In one patient the curve deteriorated clearly after reducing brace wearing time and therefore was due to non-compliance.

316

H.-R. Weiss and S. Bohr / Conservative Scoliosis Treatment

Figure 2. Surface scans of the patient who in the end was progressive due to non-compliance (Start of treatment, intermediate result and final result at the age of 19 years) without significant clinical deterioration.

4.

Discussion

In current literature there is no evidence that scoliosis surgery in patients with PraderWilli syndrome improves signs and symptoms of scoliosis during lifetime.Therefore to expose this patient population to the high risks of surgery [3] seems not to be justified. Conservative management can prevent curve progression in patients with PraderWilli syndrome and has to be regarded to be indicated primarily. On the other hand scoliosis in the case of a Prader-Willi syndrome seems to be less malignant than in other conditions when the results of conservative treatment are good at average in patients usually presenting with significant obesity.

Figure 3. Clinical pictures of the patient who in the end was progressive due to non-compliance (Start of treatment, intermediate result and final result at the age of 19 years) without significant clinical deterioration and in his last brace on the right.

H.-R. Weiss and S. Bohr / Conservative Scoliosis Treatment

317

Figure 4. 24 year old patient (to be seen on the right) next to her mother at the final follow-up. Her curve has improved significantly (see figure 1.) during the four years without brace, however performing physiotherapy regularly.

5.

Conclusions

Stabilisation of scoliosis due to Prader-Willi Syndrome is possible by means of conservative management. To expose this patient population to surgical management seems not to be justified medically.

References [1] [2] [3]

Gurd AR, Thompson TR: Scoliosis in Prader-Willi syndrome. J Pediatr Orthop. 1981;1(3):317-20. Kroonen LT, Herman M, Pizzutillo PD, Macewen GD: Prader-Willi Syndrome: clinical concerns for the orthopaedic surgeon. J Pediatr Orthop. 2006 Sep-Oct;26(5):673-9. Yamada K, Miyamoto K, Hosoe H, Mizutani M, Shimizu K: Scoliosis associated with Prader-Willi syndrome. Spine J. 2007 May-Jun;7(3):345-8.

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Chapter 9 Abstracts

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Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved.

377

Subject Index 3-D correction 116 adolescent idiopathic scoliosis 116, 133, 321–323, 326, 336, 344–346, 360 aetiology of idiopathic scoliosis 240 aetiology 340 AIS 249 arm 189 asymmetrical 221 asymmetrical growth 33 asymmetry genes 326 back morphology 29 back pain 249 balance 307 balance dysfunction 333 basicranium 360 belief propagation 161 biomechanical model 128 biomechanics 52, 90, 116, 273 body composition 344 body mass index 9 body schema 208 bone mineral 323 bone mineral density 328, 344, 345, 348 brace 299 brace management 294 brace treatment 303 buccal epithelial cells 3 calcaneus 231 calvarium 360 chondrocyte 324, 347 classification 299 cluster analysis 336 Cobb angle 151 Cobb angle improvement 337 complications 314 computer-aided measurement 151 congenital scoliosis 310 conservative treatment 281, 310 conservative treatment of idiopathic scoliosis 33

curve severity 346 cytogenetic abnormalities 3 deformity 273 diet 340 dome shaped sacrum 339 dual kriging 61 education 281 ER β 322 ergonomics 103 estrogen receptor gene 330 etiology 324, 347 evaluation 303 exercises 307, 331 experimental design 116 eye asymmetry 340 faulty posture 107 FBN3 321 femoral anteversion 37, 225 finite element model 111 finite element modeling 90 finite element simulation 85 foot 231 fusionless surgery of idiopathic scoliosis 33 gait 61 gene polymorphism 321–323, 325 genetics 330 growth 85, 273 growth hormone 346 growth plate 37, 225, 349 growth spurt 133 height loss 48 historic studies 52 human evolution 9 human torso 96 hypothalamus 197 idiopathic scoliosis 9, 22, 33, 37, 61, 72, 189, 197, 208, 225, 240, 245, 263, 281, 299, 303, 324, 325, 347–349, 371 image registration 254 image segmentation 161

378

implant configuration 111 improvement of school screening 245 in situ bending 72 in vivo 273 independent component analysis 96 indication for surgery 310 indications 314 instrumentation 128, 294 intensity image registration 96 intermembral index 189 intervertebral disc 33, 273 intervertebral efforts 61 ISIS2 65, 68, 157 juvenile idiopathic scoliosis 29 juveniles 29 kyphosis 52, 68 kyphosis angle 137 laser correlation spectroscopy 3 leg 189, 231 leptin 9, 197 literature review 331 loading 221 lordosis 231 lower limbs 90 lumbar 231 lumbar lordosis 103 machine learning 254 magnetic resonance imaging 360 marker placement 166 MATN1 325 melatonin receptor 347, 371 melatonin receptor gene 330 mesenchymal stem cell(s) 345, 371 metabolic shifts 3 mRNA 347, 348, 371 multibody dynamics 61 musculoskeletal disorders 3 natural history 133 neuro-osseous timing of maturation 208 objectivity 140 occurrence 346 optical trunk molding 22 optimization 116 osteocalcin 348 osteoneural growth 52 osteoprotegerin 345, 348

pathogenesis

9, 37, 189, 197, 208, 225, 263, 345 pathogenesis of idiopathic scoliosis 33 pattern recognition 336 pelvic balance 338 pelvis 9 physical activity 107 physical therapy 307 physiotherapy 103 pixel matching 161 polymorphism 346 posterior surgical instrumentation 337 postural control 140 posture 103 Prader-Willi syndrome 314 progression 85 psychometric assessment 332 quality and quantity of brace usage 294 quality of life 249 Quantec 337 questionnaire 249 radiography 48, 137 RANKL 345 rat model 273 reduction techniques 338 registration 161 rehabilitation 140, 331 retrospective multi-centre study 338 rib hump 65 ribcage 9 ribs vertebral rotation 263 rucksack 221 Runx2 349 sagittal deformity patterns 72 school screening 240, 245 scoliometer 337 scoliosis 52, 65, 85, 90, 111, 128, 151, 157, 161, 166, 254, 294, 307, 314, 328, 332, 333 scoliosis surgery 116 shoulder girdle 9 skeletal growth 208 skeletal maturity 48 Sox9 324 spinal length 48 spinal tension 52

379

spine

9, 37, 90, 128, 166, 189, 197, 208, 225, 231, 263 SpineCor 133 spine modeling 61 spondylolisthesis 338, 339 SRS-22 249 SRS-22r 332 staple 111 stapling of the spine 33 stature 48 style of life 107 surface topography 157 surgery 111, 314 surgery simulator 116 surgical positioning 90 susceptibility 322, 325 swimmers 340

symmetrical sympathetic nervous system thoracic growth thoracolumbar junction thoracolumbar spine tibial torsion trunk asymmetry type I collagen type X collagen ultrasound vestibular system videorasterstereography visualization vitamin D receptor volume VRS

221 197 22 72 52 37, 225 29 328, 348 349 37, 225 333 137, 140 65 323 22 137, 140

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Research into Spinal Deformities 6 P.H. Dangerfield (Ed.) IOS Press, 2008 © 2008 The authors and IOS Press. All rights reserved.

381

Author Index Ajemba, P.O. 362, 363, 375 Alchinova, I.B. 3 Aleschenko, A.V. 3 Anderson, S.I. 197 Aronsson, D.D. 273 Asher, M.A. 332, 352 Atanasio, S. 299, 303, 307, 331 Aubin, C.-É. 61, 90, 111, 116, 128, 336, 342, 356–358, 368, 376 Aujla, R.K. 9, 37, 189, 225, 263 Azeddine, B. 326, 372 Bagnall, K.M. 372 Bakaloudis, G. 289 Barbanti Brodano, G. 289 Bardakos, N. 33 Beaudoin, G. 369 Beauséjour, M. 334, 341 Bekhiche, S. 341 Berryman, F. 65, 68, 157 Berthonnaud, É. 338, 339 Bettany-Saltikov, J. 103, 221 Bohr, S. 314 Bourgin, J.-F. 22 Bowden, G. 157 Bozonnat, M.-C. 22 Breakwell, L.M. 337 Brown, C. 339 Burton, D.C. 332, 352 Burwell, R.G. 9, 37, 189, 197, 208, 225, 263, 333, 367 Calancie, B. 350 Cancel, M. 351 Cao, X.-B. 321, 322, 323 Carlson, B. 352 Chan, C.W. 361 Charles, Y.P. 22, 72 Charron, G. 369 Chen, H.-O. 321–323 Chen, W.-J. 321, 323 Chen, Z.-J. 321–323, 325 Cheng, A. 333, 367

Cheng, J.C.-Y. 325, 327, 328, 330, 333, 346, 347, 354, 360, 361, 366, 367, 370, 371, 373, 374 Cheng, T.H. 360 Cheriet, F. 269, 353, 364 Chevalier, T.L. 166 Chockalingam, N. 166, 231 Chopin, D. 338 Chow, D.H.K. 374 Chu, W.C.W. 333, 360, 366, 367 Ciazynski, D. 281 Cioni, A. 289 Clin, J. 376 Cole, A.A. 9, 37, 189, 225, 263, 337 Crandall, D. 128 Czakwari, A. 107 Czernicki, K. 107, 281 D’Amico, G. 79 D’Amico, M. 79 Dangerfield, P.H. v, 9, 37, 166, 189, 197, 208, 225, 231, 263 Dansereau, J. 90 Davies, N. 157 de Guise, J.A. 335, 365 Debanné, P. 364 Deschênes, S. 369 Detrembleur, C. 61 Di Silvestre, M. 289 Dimeglio, A. 22 Douglas, D.L. 337 Drevelle, X. 85 Driscoll, C. 90 Driscoll, M. 356 Dubois, J. 369 Dubousset, J. 85 Durdle, N.G. 96, 144, 161, 254, 362, 363, 375 Durmala, J. 107, 281 Ebermeyer, E. 85 Elobeidi, N. 137 Elsafi, A.S. 96, 144

382

Fairbank, J. Fan, H. Fisette, P. Forcier, M. Franco, A. Frascarello, M. Freeman, B.J.C.

65, 68, 157 327 61 334, 341 372 79 37, 189, 208, 225, 263, 367 Fusco, C. 303, 331 Giacomini, S. 289 Greenhalgh, A. 231 Greggi, T. 289 Grimard, G. 326, 334, 341, 351, 372 Grivas, T.B. 29, 33, 240, 245 Gum, J.L. 352 Hedden, D. 294 Heng, P.A. 333, 360, 367 Hill, D. 151, 249, 294 Hresko, T. 338 Hu, S. 339 Hu, Y.Y. 361 Huang, A. 324, 347 Hughes, C. 337 Hui, C.F.F. 361 Hung, V.W.Y. 327, 328, 354, 360 Hung, W.Y. 373 Jackson, R. 368 Jobson, M. 103 Joncas, J. 334, 341, 357 Jones, T. 359 Kadoury, S. 269, 353 Karganov, M.Y. 3 Karski, T. 194 Khlebnikova, N.N. 3 Kirby, A.S. 9, 37, 189, 225, 263 Kotwicki, T. 44 Kozhevnikva, M.I. 3 Kumar, A. 161 Kwok, R.H.K. 327, 328, 374 Labelle, H. 90, 116, 128, 269, 326, 334–336, 338, 339, 341, 343, 353, 357, 364, 365, 368, 369, 372, 376 Lacroix, G. 372 Lafon, Y. 85 Lai, S.M. 332, 352 Lalonde, N.M. 111 Lam, T.P. 327, 366, 374 Lam, W.W.M. 366 Lamarre, M.-E. 357

Larouche, G. 372 Lavy, C. 157 Le, L.H. 151 Lee, K.M. 327, 328, 330, 333, 354, 360, 361, 366, 367, 370, 373 Lee, W.T.K. 354, 374 Lenke, L. 368 Leung, K.S. 361 Li, H. 371 Li, W.-G. 322, 344, 348, 349 Lin, H. 342 Liu, X.C. 355, 359 Liu, Z. 322, 345 Lolli, F. 289 Lou, E. 151, 249, 294 Lyon, R. 355, 359 Ma, W. 345, 348, 349 Mac-Thiong, J.-M. 365 Mahood, J. 249, 294 Maier-Hennes, A. 140 Majdouline, Y. 116 Man, C.-W. 360, 370 Man, G.C.W. 333, 366, 367, 373 Marcoul, A. 22 Marcoul, M. 22 Martikos, K. 289 Maziotou, C. 29 McBride, C.A. 273 McMaster, M.E. 340 Meunier, J.E. 368 Meyer, N. 72 Mezghani, N. 335 Mihaila, D. 350 Mihas, C. 29 Miled, F. 364 Minozzi, S. 331 Miron, M.-C. 369 Moldovan, F. 351 Moreau, A. 326, 356, 372 Moreau, M. 294 Moulton, A. 9, 37, 189, 197, 225, 263 Nault, M.L. 343 Naylor, B. 337 Negrini, S. 299, 303, 307, 331 Newton, P. 368 Ng, B.K.W. 327, 328, 360, 366, 370, 373, 374 Noskin, L.A. 3 O’Brien, J.P. 245

383

O’Brien, M. 338 Ouellet, J. 326, 372 Pan, W.M. 361 Paniccia, M. 79 Pannetier, R. 111 Parent, E. 249, 294 Parent, S. 326, 334, 335, 341–343, 356, 357, 369, 372, 376 Parisini, P. 289 Parker, N. 231 Paus, T. 333, 367 Pazos, V. 364 Pelevina, I.I. 3 Petit, Y. 357 Phan, P. 335 Pitiot, A. 367 Poitras, B. 326, 372 Polak, F.J. 9, 37, 189, 225, 263 Pratt, R.K. 9, 37, 189, 225, 263 Püschel, I. 140 Pynsent, P. 65, 68, 157 Qaimkhani, S.A. 337 Qian, B. 345, 348, 349 Qin, L. 328, 354, 361 Qiu, X.-S. 321–323, 330, 346 Qiu, Y. 321–325, 327, 330, 344–349, 370, 371, 373 Quraishi, N. 157 Raison, M. 61 Ramirez, L. 254 Raso, V.J. 96, 151, 161, 254, 294, 362, 363, 375 Rivard, C.-H. 326, 372 Rivard, M. 343 Rizza, R. 355 Rodopoulos, G. 33, 240 Romano, M. 303, 307, 331 Rompré, P.H. 372 Roncoletta, P. 79 Roussouly, P. 338, 339 Roy-Beaudry, M. 334, 341, 343 Samin, J.-C. 61 Sangole, A.P. 336, 358, 368 Schumann, K. 140 Serebranyi, A.M. 3 Shannon, T.M.L. 121 Sharma, S. 337

Shi, L. 333, 360, 367 Shum, S.L.F. 374 Sin, S.W. 374 Skalli, W. 85 Stamp, M. 221 Steib, J.-P. 72 Stokes, I.A.F. 48, 273, 336, 358 Sun, G. 324, 347, 371 Sun, X. 348 Tang, N.L.-S. 325, 327, 328, 330, 346 Tassone, C. 355, 359 Ternovoy, K.S. 3 Thometz, J. 355, 359 Triandafyllopoulos, G. 29 Turgeon, I. 372 Vallasciani, M. 79 van Loon, P.J.M. 52, 170 van Rhijn, L.W. 170 Vasiliadis, E.S. 29, 33, 240, 245 Villemure, I. 111, 342, 351 Vommaro, F. 289 Wang, B. 345, 348, 349 Wang, D.S. 326, 333, 367 Wang, S. 349 Wang, W.-J. 347, 370, 371, 373 Wang, X. 128 Wang, Y. 151 Wang, Z. 365 Warren, J. 103, 221 Webb, J.K. 9, 37, 189, 225 Weiss, H.-R. 133, 137, 140, 310, 314 Wilson-MacDonald, J. 157 Wong, M.S. 374 Yeung, H.Y. 327, 328, 330, 333, 354, 360, 366, 367 Yeung, Hiu-Yan 370 Yeung, Hiu-Yen 346 Yeung, Y.H. 373 Young, M.K. 166 Yu, Y. 345, 348, 349 Zaina, F. 299, 303, 307, 331 Zewail, R. 144 Zhang, J. 151 Zhu, F. 345, 348, 349 Zhu, Z. 345, 348, 349 Ziliani, V. 307 Zubović, A. 157

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