Minority Recruitment in Cancer Genetics Studies (Community Genetics 2008)

Minority Recruitment in Cancer Genetics Studies

Guest Editors

Deborah J. Bowen, Boston, Mass. Victor Penchaszadeh, Buenos Aires

7 figures and 18 tables, 2008

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Vol. 11, No. 4, 2008

Contents

215 Testing Three Different Cancer Genetics Registry

Editorial 189 Special Issue: Enhancing Minority Recruitment into

Genetics Research Bowen, D.J. (Boston, Mass.); Penchaszadeh, V.B. (New York, N.Y./ Buenos Aires)

Recruitment Methods with Hispanic Cancer Patients and Their Family Members Previously Registered in Local Cancer Registries in Texas Ramirez, A.G.; Miller, A.R.; Gallion, K.; San Miguel de Majors, S.; Chalela, P.; García Arámburo, S. (San Antonio, Tex.) 224 Factors Associated with African Americans’

Introduction

Enrollment in a National Cancer Genetics Registry

191 Increasing Minority Participant Enrollment into a

Cancer Family Registry: The Cancer Genetics Network Bowen, D.J. (Boston, Mass.); Vu, T. (Seattle, Wash.); Kasten-Sportes, C. (Bethesda, Md.)

Skinner, C.S.; Schildkraut, J.M.; Calingaert, B.; Hoyo, C.; Crankshaw, S.S.; Fish, L. (Durham, N.C.); Susswein, L. (Chapel Hill, N.C.); Jasper, C. (Washington, D.C.); Reid, L. (Durham, N.C.) 234 Testing Targeted Approaches to Enhance Cancer

193 BRCA1 and BRCA2 Risk Perceptions among African

American Women at Increased Risk for Hereditary Breast-Ovarian Cancer Kessler, L.; Domchek, S.; Stopfer, J.; Halbert, C.H. (Philadelphia, Pa.) 201 Partnership with an African American Sorority to

Enhance Participation in Cancer Genetics Research Olsen, S.J. (Baltimore, Md.); Malvern, K.T. (Washington, D.C.); May, B.J. (Baltimore, Md.); Jenkins, I.L. (Washington, D.C.); Griffin, C.A. (Baltimore, Md.) 208 Successful Strategies for Increasing African American

Participation in Cancer Genetic Studies: Hopeful Signs for Equalizing the Benefits of Genetic Medicine Patterson, A.R.; Davis, H.; Shelby, K.; McCoy, J.; Robinson, L.D.; Rao, S.K.; Banerji, P.; Tomlinson, G.E. (Dallas, Tex.)

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Genetics Network Minority Recruitment within Asian Populations Wenzel, L. (Irvine, Calif.); Bowen, D. (Boston, Mass.); Habbal, R.; Leighton, N. (Irvine, Calif.); Vu, T. (Seattle, Wash.); Anton-Culver, H. (Irvine, Calif.) 241 Strategies and Stakeholders: Minority Recruitment in

Cancer Genetics Research James, R.D.; Yu, J.-H.; Henrikson, N.B.; Bowen, D.J.; Fullerton, S.M. (Boston, Mass.) for the Health Disparities Working Group 250 Author Index 250 Subject Index

Editorial Community Genet 2008;11:189–190 DOI: 10.1159/000116876

Special Issue: Enhancing Minority Recruitment into Genetics Research Deborah J. Bowen a Victor B. Penchaszadeh b, c a

Social and Behavioral Sciences, School of Public Health, Boston University, Boston, Mass., b Mailman School of Public Health, Columbia University, New York, N.Y., USA; c Pan American Health Organization, Buenos Aires, Argentina

Participation of families and patients from ethnic minorities in health research in general and genetics research specifically is lower than participation from Caucasian families in the US [1]. This lower participation of minorities is problematic from both a scientific and a social justice viewpoint. From a scientific standpoint, lack of participation of ethnic minorities prevents the exploration of specific ethnic differences in patterns of disease [2–4]. In turn, the lack of study of the genetic patterns of disease and risks among diverse ethnic and racial groups leads to the inability to identify differential risks among ethnic groups. Furthermore, although it is widely recognized that health disparities between ethnic groups are overwhelmingly environmental in nature (differences in socioeconomic status, education, culture, lifestyles, etc.) [5, 6], the lack of genetic studies in minorities prevents to rule out that differences in health status among ethnic groups could be due in part to genetic differences. This knowledge is critical as we move forward to apply genetic approaches to modern medicine. From a social justice standpoint, it is important to create research settings that have equitable access to participate for all persons, independent of ethnic background and other social status and structure variables. There is some evidence that people who participate in research projects, specifically clinical trials, report better health outcomes than do people who do not participate in research [7]. There are many hypotheses as to why this would be true, including (1) a selfselection bias in the groups recruited such that recruited © 2008 S. Karger AG, Basel 1422–2795/08/0114–0189$24.50/0 Fax +41 61 306 12 34 E-Mail [email protected] www.karger.com

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people are healthier than people not recruited, and (2) the high quality treatment, surveillance, and follow-up provided to participants in clinical trials versus the more variable quality provided to the general public. For these reasons, it is important to come up with methods to improve access to research participation for disadvantaged minorities. Therefore, we need to identify methods of increasing participation of ethnic minorities into genetic research projects. To date, recruitment into cancer genetics studies has mostly focused on enriched families with multiple cases of the cancer under study, often from clinical settings where genetic testing is provided [6]. Furthermore, patients recruited for those studies have been mostly Caucasian or White, with little targeted efforts to engage nonWhite participants in research. Given that minority participation in research is lagging and that knowledge on minorities is important to inform cancer prevention and care policies, the National Cancer Institute funded the Cancer Genetics Network with the task to research on minority participation in cancer studies and find methods to enhance it. The articles in this special issue of Community Genetics present a variety of approaches to enhance minority recruitment into large, populationbased studies. We hope that this collection of studies will help investigators to enhance recruitment of minority participants in their studies and that this will lead to better ways of preventing cancer.

Deborah Bowen Social and Behavioral Sciences, Boston University 715 Albany Street T2W Boston, MA 02118 (USA) Tel. +1 617 638 5205, Fax +1 617 638 4483, E-Mail [email protected]

References 1 Hughes C, Peterson S, Ramirez A, Gallion K, McDonald P, Skinner C, Bowen DJ: Minority recruitment in hereditary breast cancer research. Cancer Epidemiol Biomarkers Prev 2004;13:1146–1155. 2 Burchard EG, Ziv E, Coyle N, Gomez SL, Tang H, Karter AJ, Mountain JL, Perez-Stable EJ, Sheppard D, Risch N: The importance of race and ethnic background in biomedical research and clinical practice. N Engl J Med 2003;348:1170–1175.

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3 Cooper RS, Kaufman J, Ward R: Race and genomics. New Engl J Med 2003; 348: 1166– 1170. 4 Krieger N, et al: Painting a truer picture of US socioeconomic and racial/ethnic health inequalities: the Public Health Disparities Geocoding Project. Am J Public Health 2005;95: 312–323. 5 Institute of Medicine: Unequal Treatment: Confronting Racial and Ethnic Disparities in Healthcare. Washington, National Academy of Press, 2003.

Community Genet 2008;11:189–190

6 Sankar P, Cho MK, Condit CM, Hunt LM, Koenig B, Marshall P, Soo-Jin Lee S, Spicer P: Genetic research and health disparities. JAMA 2004;291:2985–2989. 7 Kandzari DE, Roe MT, Chen AY, Lytle BL, Pollack CV, Harrington RA, Ohman EM, Gibler WB, Peterson ED: Influence of clinical trial enrollment on the quality of care and outcomes for patients with non-ST-segment elevation acute coronary syndromes. Am Heart J 2005;149:474–481.

Bowen /Penchaszadeh

Introduction Community Genet 2008;11:191–192 DOI: 10.1159/000116877

Increasing Minority Participant Enrollment into a Cancer Family Registry: The Cancer Genetics Network Deborah J. Bowen d Thuy Vu a, b Carol Kasten-Sportes c a Fred Hutchinson Cancer Research Center and b University of Washington, Seattle, Wash.; c National Cancer Institute, Bethesda, Md.; d Social and Behavioral Sciences, School of Public Health, Boston University, Boston, Mass., USA

In 1998, the National Cancer Institute funded an innovative national Cancer Genetics Network (CGN) which provides participating researchers access to a breadth of research data not currently available to most individual cancer genetics programs. The CGN is composed of eight centers around the nation which are linked to a central informatics center. The Network supports collaborations to investigate the genetic basis of cancer susceptibility, explore mechanisms to integrate this new knowledge into medical practice, and identify ways to address associated psychosocial, ethical, legal, and public health issues [1]. As of May 2002, the CGN contained data on 15,007 participants and 241,948 family members. The majority of CGN participants were of Non-Hispanic White/Caucasian ethnicity (90%), with few numbers of Hispanic (4%), Black (3%), Asian (1%), and other ethnicities (2%). These participation figures did not match those of the general public, nor did they match the catchment areas of the participating sites in the CGN. The investigators and NIH staff reviewed the disparities in recruitment yield 4 years after the registry funding period began, and decided to take action to improve recruitment of minority participants. The investigators decided to conduct research into minority recruitment, focusing on ways to enhance ethnic minority participation into the registry. The sites were charged with two goals: to identify new recruitment sources appropriate to the locale and eligibility requirements of each site, and/or to © 2008 S. Karger AG, Basel 1422–2795/08/0114–0191$24.50/0 Fax +41 61 306 12 34 E-Mail [email protected] www.karger.com

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design and implement studies at each site to test the usefulness of new recruitment strategies, processes, or incentives. A published paper came out of this initial effort to review the literature on this topic [2], as well as a series of ideas and processes to test within the CGN sites. The research team formed working groups, based on scientific expertise located at each center, populations existing in the catchment area of each site, and interest in participating in an effort in minority recruitment. Each of the working groups focused on a package of a different ethnic minority population and a different strategy. The centers at Duke and Georgetown Universities conducted a randomized trial of a recruitment brochure in which the content for potential African American enrollees to the CGN was targeted and enhanced based on formative research with the targeted population. Investigators at the University of Pennsylvania tested the offering of personal risk feedback as an incentive to join CGN, adding questions to the standard CGN survey to capture reactions to the feedback. Efforts at Johns Hopkins focused on working with African American sororities as community contacts to enhance enrollment into the network. The group at the University of Texas conducted a randomized trial to test the effects of a targeted print media product, a magazine, to enhance recruitment of Hispanic participants into the CGN. The collaborative study conducted by investigators at both the University of California Irvine and the Fred Hutchinson Cancer Research Center Deborah Bowen Social and Behavioral Sciences Boston University, 715 Albany Street T2W Boston, MA 02118 (USA) Tel. +1 617 638 5205, Fax +1 617 638 4483, E-Mail [email protected]

was a randomized trial of a pan-Asian message of welcome to the CGN to increase Asian recruitment. The papers in this focused issue present the results of these efforts by working group. Each manuscript describes an enhanced recruitment process or procedure and presents data on its effectiveness. In addition, most of the project teams collected data outside of the standard CGN enrollment data to enable a greater understanding of the recruitment process for minority individuals. Overall enrollment of minority participants into the CGN was enhanced by these minority recruitment efforts. The enrollment of Black, Hispanic, and Asian participants over the period of the minority recruitment efforts increased steadily, from 1,315 participants to 1,942

References

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participants. The efforts of the specific studies focused on minority participants drove this change. This collaborative activity represented our first attempt to identify issues that would produce lower enrollment and to enhance the attractiveness of participating in a cancer registry for minority individuals. We hope that the data collected and presented here will be useful to improving recruitment yields in future research projects of this type. We also find this type of multidisciplinary collaboration useful in creating a setting where issues of race, recruitment, and scientific discussions can occur and can lead to new data efforts. We hope to continue this type of work in the future.

1 Anton-Culver H, Ziogas A, Bowen D, Finkelstein D, Griffin C, Hanson J, Isaacs C, Kasten-Sportes C, Mineau G, Nadkarni P, Rimer B, Schildkraut J, Strong L, Weber B, Winn D, Hiatt R, Nayfield S: The Cancer Genetics Network: recruitment results and pilot studies. Community Genet 2003;6:171–177.

Community Genet 2008;11:191–192

2 Hughes C, Peterson S, Ramirez A, Gallion K, McDonald P, Skinner C, Bowen DJ: Minority recruitment in hereditary breast cancer research. Cancer Epidemiol Biomarkers Prev 2004;13:1146–1155.

Bowen /Vu /Kasten-Sportes

Community Genet 2008;11:193–200 DOI: 10.1159/000116879

BRCA1 and BRCA2 Risk Perceptions among African American Women at Increased Risk for Hereditary Breast-Ovarian Cancer Lisa Kessler a Susan Domchek b, c Jill Stopfer c Chanita Hughes Halbert a, c, d Departments of a Psychiatry and b Medicine, c Abramson Cancer Center, and d Leonard Davis Institute of Health Economics, University of Pennsylvania, Philadelphia, Pa., USA

Key Words BRCA1/2 mutation ⴢ Risk perception ⴢ African American women

Abstract Objectives: To describe BRCA1 or BRCA2 (BRCA1/2) risk perceptions among African American women at increased risk for hereditary breast-ovarian cancer and to identify factors having independent associations with these perceptions. Methods: Risk perceptions were evaluated by self-report during a structured telephone interview among African American women (n = 162) at increased risk for hereditary cancer who were recruited from oncology clinics, general medical practices, and community oncology resources. Results: The majority of women (75%) believed that it was likely that they had a BRCA1/2 mutation. Women ages 50 and younger and those with greater cancer-specific worry were most likely to believe that they had a BRCA1/2 mutation. Conclusions: Although BRCA1/2 risk perceptions may be consistent with objective risk levels among African American women, discussion about the basis of risk perceptions may enhance provision of genetic counseling and testing in this population. Copyright © 2008 S. Karger AG, Basel

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Introduction

Similar to Caucasian individuals without Ashkenazi Jewish ancestry, epidemiological studies have shown that the prevalence of BRCA1 and BRCA2 (BRCA1/2) mutations ranges between 16–21% among African American women who have a personal or family history of breast and/or ovarian cancer that is suggestive of hereditary disease [1–3]. Genetic counseling and testing are now being integrated into the clinical management of women who have an increased risk of having a BRCA1/2 mutation; within these programs, pedigree-based risk assessments may be completed to provide information about the likelihood that a risk-conferring mutation is present [4, 5]. However, interest in these services may be limited among African Americans. Recent studies have shown that African American women were significantly less likely than Caucasian women to participate in genetic counseling offered in a clinical setting [6] and about 50% of African American women participated in genetic counseling offered as part of a research protocol [7]. Moreover, rates of BRCA1/2 test result acceptance may be as low as 21% among African American women because of cancer-related stressors [8]. Limited African American participation in genetic counseling and testing programs may have Chanita Hughes Halbert, PhD University of Pennsylvania 3535 Market Street, Suite 4100 Philadelphia, PA 19104 (USA) Tel. +1 215 747 7144, Fax +1 215 746 7140, E-Mail [email protected]

implications for recruitment into other types of genetic research (e.g. registry studies) since high-risk cancer clinics may be used for subject recruitment [9]. Regardless of the setting in which genetic counseling and testing are offered, interest in utilizing these services may be influenced by perceived risk of having a BRCA1/2 mutation. African Americans who believed that they were at risk for having a BRCA1/2 mutation were most likely to be interested in genetic testing [10, 11]. However, most African Americans at increased risk for hereditary breast cancer may not believe that they are at risk for having a BRCA1/2 mutation. Only about 20–30% of African Americans from a family with a known risk-conferring BRCA1/2 mutation believed that they were at risk for having a BRCA1/2 mutation [10]. In contrast, McInerney-Leo et al. [12] found that 97% of individuals from a sample composed primarily of Caucasian families with known BRCA1/2 mutations believed that it was likely they carried a mutation. Previous research has also shown that African American women are more likely than Caucasian women to base cancer-related risk perceptions on subjective experiences. For example, having a later age at first live birth and having a relative diagnosed with more advanced stage breast cancer were among the predictors of heightened risk perceptions among Caucasian women; however, only personal concerns about developing breast cancer and moderate levels of worry about a relative recently diagnosed with breast cancer were associated significantly with heightened perceived risk among African American women [13]. Similarly, Salant et al. [14] found that risk perceptions were conceptualized in terms of personal experiences with breast cancer in a sample composed primarily of African American women. Although the probability of having a BRCA1/2 mutation is based on objective risk factors such as personal and family history of cancer [15–17], these factors may be less important to BRCA1/2 risk perceptions relative to cancer-related worries among African American women. However, empirical data are not available on factors that are associated with BRCA1/2 risk perceptions among African American women at increased risk for hereditary disease. In 1998, the National Cancer Institute funded a familial cancer registry called the national Cancer Genetics Network (CGN) to provide participating researchers access to a breadth of research data not then available to most individual cancer genetics programs. The CGN is comprised of eight main centers around the nation that are linked to a central informatics center. The Network supports collaborations to investigate the genetic basis of cancer susceptibility, explore mechanisms to integrate 194

Community Genet 2008;11:193–200

this new knowledge into medical practice, and identify ways to address associated psychosocial, ethical, legal, and public health issues [18]. Because minority participation rates lagged behind those of Caucasian families [Bowen, Vu and Kasten-Sportes, this issue, pp. 191–192], the CGN investigators initiated a program (1) to increase minority enrollment in the CGN and (2) to contribute to the scientific knowledge about enhancement of minority participation in genetics studies. This study presents data from one of the projects in that effort. Specifically, this study evaluated perceived risk of having a BRCA1/2 mutation among African American women at increased risk for hereditary breast and ovarian cancer. We focused on perceived risk, or one’s subjective assessment of the likelihood of having a mutation in this report because of its importance to participation in genetic counseling and testing [19] and the subsequent implications of participation in genetic counseling on recruitment into cancer genetics registries. Since risk perception is an important component of health behavior theory (e.g. Health Belief Model) [20] that can be used to personalize cancer risk information, data on BRCA1/2 risk perceptions and factors that are associated with perceived risk among African American women prior to participation in genetic counseling and testing is also important for developing more effective genetic counseling protocols for this population. Based on prior research showing that cancer-related risk perceptions may be influenced by worries about developing cancer [13, 21], we evaluated the independent associations between BRCA1/2 risk perceptions and cancer-specific worry and objective risk factors for hereditary breast-ovarian cancer in the present study.

Materials and Methods Study Population Participants were African American women (n = 162) who were at increased risk for having a BRCA1/2 mutation. To be eligible for participation in the study, women had to self-identify as being African American or Black. Women also had to have at least a 5–10% prior probability of having a BRCA1/2 mutation based on their personal and family history of breast and/or ovarian cancer to be eligible for study participation since this is the minimum criteria for clinical genetic testing [22]. Women who had previously received genetic counseling or clinical testing for BRCA1/2 mutations were excluded from study participation. Women were recruited into the study from February 2003 through July 2005.

Kessler /Domchek /Stopfer /Halbert

Procedures Women were recruited into the study through a referral network that included oncology clinics, general medical practices, and community oncology resources (e.g. breast cancer support groups, health fairs) located in Philadelphia. Women recruited from clinical facilities were given brochures and flyers that described the study (e.g. a new research program that provided counseling and education to eligible African American women) by physicians and clinic staff. This information was distributed by research staff at community oncology resources. Women who were interested in learning more about the study completed a referral form that collected race, contact information, date of birth, and personal and family history of breast and ovarian cancer. Referral forms were reviewed by the study genetic counseling (L.K.) to determine eligibility. Eligible women were mailed an invitation letter that described the purpose of the study and the procedures involved in participation. Women who did not opt out of enrollment by returning a reply card were contacted by telephone for study enrollment and to complete the baseline telephone interview. Some women (n = 25) provided a blood sample as part of a separate epidemiological study to understand genetic risk factors for breast cancer in African American women before their enrollment in this study. However, these women did not receive genetic counseling and risk assessment for inherited breast-ovarian cancer risk, and clinical genetic testing for BRCA1/2 mutations was not performed prior to their participation in the present study. Further, participation in the epidemiological study did not influence decisions about enrollment in this study [7] or perceived risk of having a BRCA1/2 mutation (␹2 = 1.36, p = 0.24). Among the eligible women who were identified and invited to participate in the study, 62% enrolled and completed the baseline telephone interview [7]. Study enrollment and the baseline telephone interview were completed by a trained telephone interviewer from the University of Pennsylvania and took about 40 min to complete. The baseline was a structured survey that assessed sociodemographic characteristics, cancer-specific worry, and BRCA1/2 risk perceptions. Prior to asking about perceived risk of having a BRCA1/2 mutation, women were read a brief, standardized script that described the characteristics of hereditary breast-ovarian cancer, BRCA1/2 susceptibility genes, and information about the availability of genetic testing. At the end of the baseline, women were invited to participate in a genetic counseling session. The present report focuses on data collected during the baseline telephone interview prior to participation in genetic counseling. Measures Predictor Variables Sociodemographic Characteristics. Household income, marital status, education level, and employment status were obtained during the baseline telephone interview. These variables were recoded into dichotomous variables based on the frequency of responses (married versus not married, employed versus not employed, some college/college graduate versus high school graduate or less, household income of ^USD 35,000 versus 1USD 35,000. Objective Risk Factors. Objective risk factors for having a BRCA1/2 mutation were evaluated by self-report in terms of age, personal history of breast and/or ovarian cancer, and family history of disease. Specifically, women’s current age was categorized as age 50 or younger or older than age 50. For family history of cancer, we calculated the total number of first-, second-, and

BRCA1/2 Risk Perceptions in African Americans

third-degree relatives diagnosed with breast and/or ovarian cancer because it is standard practice to construct a three-generation pedigree for genetic counseling and this information is used to determine if one’s family history of cancer is suggestive of hereditary disease. Participants were categorized as having two or more affected relatives or less than two affected relatives. In addition to evaluating objective risk factors individually, we also evaluated overall risk in terms of prior probability of having a BRCA1/2 mutation. Prior probability of having a BRCA1/2 mutation was estimated based on the participant’s personal and family history of cancer using risk estimation models (e.g. BRCAPro) and empiric data from prior reports [22–24]. Participants were categorized as being at high (610%) or moderate (5%) risk for having a BRCA1/2 mutation. Cancer-Specific Worry. We used three Likert-style items to evaluate worry about developing breast cancer (see table 2). Specifically, women were asked how often they thought about their chances of developing breast cancer and how often these thoughts impacted their mood during the past month. In addition, women were asked how often thoughts about their risk of developing breast cancer impacted their ability to perform their daily activities. Women who had a personal history of breast cancer were asked to indicate how often they thought about their chances of developing breast cancer again, how often thoughts about developing breast cancer again impacted their mood, and how often thoughts about their risk of developing breast cancer again impacted their ability to perform their daily activities. Response options for these items were: 1 = not at all or rarely, 2 = sometimes, 3 = often, 4 = a lot. These items have been used to measure cancerspecific worry in a number of prior studies [25–27]. We recoded these items into dichotomous variables (not at all versus sometimes, often, a lot) based on the distribution of responses. Outcome Variable BRCA1/2 Risk Perception. We used one Likert-style item to evaluate perceived risk of having a BRCA1/2 mutation. Specifically, women were asked to indicate how likely it was that they had an altered breast-ovarian cancer gene (1 = not at all likely, 2 = somewhat likely, 3 = very likely, 4 = definitely). This item has been validated in previous research on interest in genetic testing [28] and has been used in prior research on education and counseling for hereditary breast cancer among African American women [11]. Data Analysis We first generated frequencies to characterize participants in terms of sociodemographic characteristics, objective risk factors, cancer-specific worry, and BRCA1/2 risk perceptions. We then conducted bivariate analyses to evaluate the relationship between BRCA1/2 risk perceptions and sociodemographic characteristics, objective risk factors, and cancer-specific worry. BRCA1/2 risk perceptions were recoded into a dichotomous variable (not at all likely versus somewhat likely, very likely, and definitely) for these analyses and we used ␹2 tests of association to evaluate the relationship between perceived risk and predictor variables. We then conducted multivariate logistic regression analysis to identify factors having independent associations with BRCA1/2 risk perceptions. Variables that had a bivariate association of p ! 0.10 with perceived risk were included in the logistic regression analysis.

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Table 1. Sample characteristics (n = 162)

Variable Marital status

Level

not married married Education level ≥some college ≤high school Employment status employed not employed Income level >USD 35,000 ≤USD 35,000 Age ≤50 >50 Cancer status affected unaffected Family history of cancer ≥2 relatives <2 relatives BRCA1/2 prior probability high moderate

Table 2. Factors associated with BRCA1/2 risk perceptions

n

%

112 50 110 52 106 56 77 85 102 60 100 62 99 63 83 79

69 31 68 32 65 35 47 53 63 37 62 38 61 39 51 49

Variable

Level

Sociodemographic characteristics Marital status not married married Education level ≥some college ≤high school Employment status employed not employed Income level >USD 35,000 ≤USD 35,000 Objective risk factors Age Cancer status Family history of cancer BRCA1/2 prior probability

Results

Sample Characteristics As shown in table 1, most participants were not married (69%), were employed (65%), and had some college education or were college graduates (68%). In terms of objective risk factors, most women had two or more relatives affected with breast and/or ovarian cancer (61%) and were at high risk for having a BRCA1/2 mutation (51%). In addition, most women had a personal history of breast and/or ovarian cancer (62%) and the majority of women affected with breast cancer had been diagnosed within the past 5 years and were short-term survivors. There were no differences between affected and unaffected women in terms of sociodemographic factors (e.g. income ␹2 = 0.02, p = 0.88; education ␹2 = 2.88, p = 0.09; marital status ␹2 = 2.10, p = 0.15, or employment status ␹2 = 0.68, p = 0.41). The mean (SD) age of participants was 46 (11.7); most participants (63%) were ages 50 or younger. Descriptive Information on BRCA1/2 Risk Perceptions Overall, most women (75%) believed that they were at risk for having a BRCA1/2 mutation. For example, 51% of participants reported that it was somewhat likely that they had an altered breast-ovarian cancer gene, 15% reported that it was very likely that they had an altered breast-ovarian cancer gene, and 8% of participants reported that they definitely had an altered breast-ovarian cancer gene. Only 25% of participants reported that it was 196

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Cancer-specific worry Thoughts about chances of developing breast cancer1 Impact of thoughts about breast cancer on mood2 Impact of thoughts about breast cancer on daily activities3

Likely have ␹2 a BRCA1/2 mutation, %

71 82 77 69 75 73 82 68

2.04 1.21 0.10 3.94*

≤50 >50 affected unaffected ≥2 relatives <2 relatives high moderate

83 60 71 81 78 70 77 72

10.88***

≥sometimes not at all ≥sometimes not at all ≥sometimes not at all

86 58 88 67 86 72

15.94***

1.88 1.28 0.53

9.38** 2.18

* p < 0.05; ** p < 0.01; *** p < 0.001. 1 Women were asked: ‘During the past month, how often have you thought about your own chances of getting breast cancer (again)?’ 2 Women were asked: ‘During the past month, how often have thoughts about your chances of getting breast cancer (again) affected your mood?’ 3 Women were asked: ‘During the past month, how often have thoughts about your chances of getting breast cancer (again) affected your ability to perform your daily activities?’

not at all likely that they had an altered breast-ovarian cancer gene. Bivariate Analysis of BRCA1/2 Risk Perceptions The results of the bivariate analysis of BRCA1/2 risk perceptions are provided in table 2. Of the sociodemographic characteristics, only income was associated significantly with perceived risk. Women with incomes greater than USD 35,000 were significantly more likely to believe that it was likely that they had a BRCA1/2 mutaKessler /Domchek /Stopfer /Halbert

Table 3. Multivariate regression model of BRCA1/2 risk perceptions

Variable

Level

OR

95% CI

Age

≤50 >50 (reference) >USD 35,000 ≤USD 35,000 (reference) ≥sometimes not at all (reference) ≥sometimes not at all (reference)

2.54 1.00 2.10 1.00 2.76 1.00 1.70 1.00

1.15–5.63**

Income Thoughts about chances of developing breast cancer Impact of thoughts about breast cancer on mood

0.94–4.66* 1.10–6.86** 0.56–5.13

* p < 0.10; ** p < 0.05.

tion compared to women with lower incomes. Of the objective risk factors, only age was associated significantly with BRCA1/2 risk perception. Women ages 50 and younger were significantly more likely to believe that it was likely that they had a BRCA1/2 mutation compared to women older than age 50. Women with greater cancerrelated worries were also most likely to believe that it was likely that they had a BRCA1/2 mutation. Specifically, women with more frequent thoughts about their chances of developing breast cancer and those whose thoughts about their chances of developing breast cancer had more of an impact on their mood were most likely to believe that they had a BRCA1/2 mutation. Marital status, education, employment status, BRCA1/2 prior probability, cancer status, family history of cancer, and the impact of thoughts about breast cancer on one’s daily activities were not associated significantly with perceived risk of having a BRCA1/2 mutation. Multivariate Regression Model of BRCA1/2 Risk Perceptions The results of the multivariate logistic regression model of perceived risk of having a BRCA1/2 mutation are provided in table 3. Variables that had a bivariate association of p ! 0.10 with perceived risk (age, income, thoughts about one’s chances of developing breast cancer, impact of worries on mood) were included in the regression model. Only age and thoughts about one’s chances of developing breast cancer had a significant independent association with perceived risk of having a BRCA1/2 mutation. Women aged 50 and younger were about three times more likely than older women to believe that they had BRCA1/2 mutation (OR = 2.59, 95% CI = 1.15, 5.63, p = 0.02). Similarly, women who had more frequent thoughts about their chances of developing breast cancer were about three times more likely than women with less BRCA1/2 Risk Perceptions in African Americans

frequent thoughts to believe that they had mutation (OR = 2.76, 95% CI = 1.11, 6.86, p = 0.03). Income level and the impact of thoughts about one’s chances of developing breast cancer on mood were not associated significantly with perceived risk of having a BRCA1/2 mutation.

Discussion

Although perceived risk of having a BRCA1/2 mutation is important to interest in participating in genetic counseling among African American women [10, 11], few studies have evaluated factors that are associated with risk perceptions among African American women at increased risk for hereditary breast-ovarian cancer. Prior studies have shown that women with a family history of cancer may overestimate their risk of developing disease [29–31]; however, our results suggest that for the most part, perceived risk of having a BRCA1/2 risk mutation may be consistent with one’s objective risk among African American women. All women included in this study had a personal or family history of breast and/or ovarian cancer that was suggestive of hereditary disease and had a 5–10% risk of having a BRCA1/2 mutation. Consistent with this, the majority of participants (75%) believed that it was likely that they had a BRCA1/2 mutation. We found that women who were younger in age were most likely to believe that they had a BRCA1/2 mutation. Age at cancer diagnosis is an important risk factor for BRCA1/2 mutations [15, 17, 22, 32]; prior reports have shown that the incidence of early onset breast cancer is greater among African American women relative to white women [33–35]. It is possible that women ages 50 and younger were more likely than older women to believe that they have a BRCA1/2 mutation because they are at Community Genet 2008;11:193–200

197

the age when other family members were diagnosed with disease. Consistent with previous research [13, 36, 37], we found that greater levels of cancer-specific worry were associated significantly with perceived risk of having a BRCA1/2 mutation. However, it is interesting to note that not all aspects of cancer-specific worry were associated with BRCA1/2 risk perceptions; only thoughts about one’s chances of developing breast cancer had a significant independent association with BRCA1/2 risk perceptions. Specifically, women with more frequent thoughts about their chances of developing breast cancer were most likely to believe that they had a BRCA1/2 mutation. It is important to note, however, that BRCA1/2 risk perceptions were not associated with prior probability of having a mutation. Further, even though some women had a personal history of cancer, cancer status and family history of disease cancer were not associated significantly with risk perceptions. Previous research has shown that knowledge about risk factors for breast cancer is limited among African American women [38]. Thus, the lack of association between objective risk factors for BRCA1/2 mutations and perceived risk may be due to limited knowledge about breast cancer genetics among African American women [10, 38, 39]. Women who are not knowledgeable about the inheritance of BRCA1/2 genes may also have limited knowledge about factors (e.g. personal history of cancer, family history of disease) that contribute to the likelihood of having a BRCA1/2 mutation. In considering the results of this study, several limitations should be considered. First, we used one Likert-style item to evaluate subjective risk of having a BRCA1/2 mutation. While subjective assessment of risk may be difficult to quantify, the item we used had acceptable face validity and has been used in previous research on hereditary breast cancer among African American women [11]. Further, other types of measures of cancer-related risk perceptions (e.g. percentages) have not been shown to be superior to frequency measures in all cases [37, 40], and single item frequency measures are often used to evaluate cancer-related risk perceptions among African American women [13, 41]. Since our results are cross-sectional and based only on African American women, it was not possible for us to compare ethnic and racial groups in terms of BRCA1/2 risk perceptions. However, our sample was similar to other populations at risk for hereditary breastovarian cancer in terms of sociodemographic factors [10, 42, 43]. Nonetheless, additional research with more detailed measures of sociodemographic factors is needed to evaluate racial differences in risk perceptions among 198

Community Genet 2008;11:193–200

women at increased risk for hereditary disease. An additional limitation is that we did not evaluate the relationship between BRCA1/2 risk perceptions and behavioral outcomes (e.g. participation in genetic counseling and testing or recruitment variables such as decisions about study enrollment) in this report. However, the results of the present study have several important implications for genetic counseling protocols targeted to African American women and recruitment of this population into cancer genetics research. Qualitative and quantitative data on prior probabilities of having a BRCA1/2 mutation may be provided to women as part of pretest education and counseling for inherited breast-ovarian cancer susceptibility, especially in research settings [4, 5]. The results of the present study underscore the importance of inquiring about the basis of BRCA1/2 risk perceptions among African American women during education and counseling about hereditary breast cancer and genetic testing. Asking women if they think that they have a deleterious BRCA1/2 mutation and the basis for these perceptions may facilitate discussions about breast cancer risk and the implications of BRCA1/2 mutation status (e.g. carrier, noncarrier, ambiguous) on cancer risk estimates. When asking about the basis for BRCA1/2 risk perceptions, it will be important to do so in a sensitive manner so that women will feel comfortable talking about beliefs that may not fit within biomedical models of disease. Previous research has shown that folk beliefs (e.g. cancer is caused by a blow to the breast) about breast cancer may be common among African American women [44]; open discussions about beliefs surrounding breast cancer and hereditary disease may be an opportunity to address misconceptions about the causes of disease and increase knowledge about breast cancer genetics. Several studies have shown that obtaining cancer risk information may be an important motivation for genetic testing for BRCA1/2 mutations among African American women [11, 38, 39, 45]. Despite this, participation in genetic counseling and testing research protocols may be limited among African American women [6–8]. Limited awareness about genetic testing for inherited disease [38] and the opportunity to receive risk information about BRCA1/2 mutations and breast and ovarian cancer may contribute to low participation rates among African American women. Recent work has emphasized the importance of awareness about study procedures on minority participation in cancer research [46]. Increasing awareness about the specific services that may be provided as part of genetic counseling and testing research Kessler /Domchek /Stopfer /Halbert

protocols may be one strategy for enhancing African American participation in these types of studies. It is important to recognize that low rates of African American participation in genetic counseling and testing programs may also translate into low rates of African American participation in cancer genetics registries since these clinics are used frequently to recruit participants for hereditary breast cancer studies [9] and may also be used to recruit participants for cancer registry studies. Thus, investigators may need to use alternative strategies to recruit African American women to participate in cancer genetic registries. Oncology settings (e.g. mammography and oncology clinics) were the most effective for recruiting African American women to participate in genetic counseling research for BRCA1/2 mutations [7]. However, results from prior studies suggest that cancerrelated risk perceptions may contribute to interest in participating in genetic testing [10, 11, 19] regardless of the recruitment setting. Risk perceptions have been used to personalize cancer risk information [20]; perceived risk of having a BRCA1/2 mutation may be important for per-

sonalizing recruitment messages and protocols to enhance African American participation in cancer genetics research. Additional research is needed to evaluate the relationship between risk perceptions and enrollment in cancer genetics research to facilitate the development of recruitment messages and protocols that address the beliefs of African American women. Future studies are also needed to evaluate changes in BRCA1/2 risk perceptions following participation in genetic counseling and testing.

Acknowledgements This research was supported by Department of Defense grant No. DAMD17-00-1-0262. We would like to thank Kiyona Brewster, Aliya Collier, ChaChira Smith, and Benita Weathers for data collection and management. We would also like to acknowledge Bridget Callaghan for assistance with manuscript preparation. We are very grateful to all of the women who participated in this study.

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10 Kinney AY, Croyle RT, Dudley WN, Bailey CA, Pelias MK, Neuhausen SL: Knowledge, attitudes, and interest in breast-ovarian cancer gene testing: a survey of a large AfricanAmerican kindred with a BRCA1 mutation. Prev Med 2001;33:543–551. 11 Kessler L, Collier A, Brewster K, Smith C, Weathers B, Wileyto EP, Halbert CH: Attitudes about genetic testing and genetic testing intentions in African American women at increased risk for hereditary breast cancer. Genet Med 2005;7:230–238. 12 McInerney-Leo A, Hadley D, Kase RG, Giambarresi TR, Struewing JR, Biesecker BB: BRCA1/2 testing in hereditary breast and ovarian cancer families III: risk perception and screening. Am J Med Genet 2006; 140A:2198–2206. 13 Hughes C, Lerman C, Lustbader E: Ethnic differences in risk perception among women at increased risk for breast cancer. Breast Cancer Res Treat 1996;40:25–35. 14 Salant T, Ganschow PS, Olopade OI, Lauderdale DS: Why take it if you don’t have anything? Breast cancer risk perceptions and prevention choices at a public hospital. J Gen Intern Med 2006;21:779–785. 15 Berry DA, Parmigiani G, Sanchez J, Schildkraut J, Winer E: Probability of carrying a mutation of breast-ovarian cancer gene BRCA1 based on family history. J Natl Cancer Inst 1997;89:227–238.

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37 Katapodi MC, Lee KA, Facione NC, Dodd MJ: Predictors of perceived breast cancer risk and the relation between perceived risk and breast cancer screening: a meta-analytic review. Prev Med 2004;38:388–402. 38 Hughes C, Gomez-Caminero A, Benkendorf J, Kerner J, Isaacs C, Barter J, Lerman C: Ethnic differences in knowledge and attitudes about BRCA1 testing in women at increased risk. Patient Educ Couns 1997; 32:51–62. 39 Donovan KA, Tucker DC: Knowledge about genetic risk for breast cancer and perceptions of genetic testing in a sociodemographically diverse sample. J Behav Med 2000; 23: 15–36. 40 Schapira MM, Davids SL, McAuliffe TL, Nattinger AB: Agreement between scales in the measurement of breast cancer risk perceptions. Risk Anal 2004;24:665–673. 41 Lipkus IM, Iden D, Terrenoire J, Feaganes JR: Relationships among breast cancer concern, risk perceptions, and interest in genetic testing for breast cancer susceptibility among African-American women with and without a family history of breast cancer. Cancer Epidemiol Biomarkers Prev 1999; 8:533–539. 42 Lerman C, Hughes C, Benkendorf JL, Biesecker B, Kerner J, Willison J, Eads N, Hadley D, Lynch J: Racial differences in testing motivation and psychological distress following pre-test education for BRCA1 gene testing. Cancer Epidemiol Biomarkers Prev 1999; 8: 361–367. 43 Halbert CH, Schwartz MD, Wenzel L, Narod S, Peshkin BN, Cella D, Lerman C: Predictors of cognitive appraisals following genetic testing for BRCA1 and BRCA2 mutations. J Behav Med 2004;27:373–392. 44 Lannin DR, Mathews HF, Mitchell J, Swanson MS, Swanson FH, Edwards MS: Influence of socioeconomic and cultural factors on racial differences in late-stage presentation of breast cancer. JAMA 1998;279:1801– 1807. 45 Thompson HS, Valdimarsdottir HB, Duteau-Buck C, Guevarra J, Bovbjerg DH, Richmond-Avellaneda C, Amarel D, Godfrey D, Brown K, Offit K: Psychosocial predictors of BRCA counseling and testing decisions among urban African-American women. Cancer Epidemiol Biomarkers Prev 2002;11:1579–1585. 46 Brown BA, Long HL, Gould H, Weitz T, Milliken N: A conceptual model for the recruitment of diverse women into research studies. J Womens Health Gend Based Med 2000; 9: 625–632.

Kessler /Domchek /Stopfer /Halbert

Community Genet 2008;11:201–207 DOI: 10.1159/000116880

Partnership with an African American Sorority to Enhance Participation in Cancer Genetics Research Sharon J. Olsen a Kathryn T. Malvern b Betty J. May a Issie L. Jenkins b Constance A. Griffin a a b

Mid-Atlantic Cancer Genetics Network, the Johns Hopkins University, Baltimore, Md., and Zeta Phi Beta Sorority, Inc., National Educational Foundation, Washington, D.C., USA

Key Words Cancer ⴢ Genetics ⴢ African American ⴢ Recruitment ⴢ Research ⴢ Cancer Genetics Network

Abstract Background/Aims: Reduced minority participation in clinical research challenges researchers to consider novel recruitment modalities. This study describes a formal partnership between the National Educational Foundation of the Zeta Phi Beta Sorority and the Mid-Atlantic Cancer Genetics Network. The goal was to enhance awareness about inherited breast cancer and to increase enrollment in the national Cancer Genetics Network. Methods: In this descriptive, pilot study, two recruitment strategies across four states were undertaken: an onsite educational session at four Annual State Leadership Conferences and a 2-tiered direct mail campaign to the sorority membership. Results: Recruitment methods targeted over 1,200 well-educated African American women. Of the 279 attendees at the state conference educational sessions, only 3 women meeting the high risk eligibility requirement enrolled. Direct mail recruitment elicited 24 eligible women. Lessons learned are described. Conclusion: Despite low accrual, the partnership laid a foundation for broader collaboration with the Zeta Phi Beta Sorority. In the future, collaboration with minority sororities and fraternities as part of standard registry recruitment should be explored. Copyright © 2008 S. Karger AG, Basel

© 2008 S. Karger AG, Basel 1422–2795/08/0114–0201$24.50/0 Fax +41 61 306 12 34 E-Mail [email protected] www.karger.com

Accessible online at: www.karger.com/cmg

Introduction

Proportionate representation of study participants by ethnic/racial group is a requirement of federally funded clinical research [1]. To understand the prevalence and relevance of genetic variance within and between populations it is important that the continuum of ethnic/racial diversity be represented in cancer genetic clinical trials. Studies have generally found that minority participation in clinical research, particularly among African Americans, is low [2–7]. Among these, recruitment varies from 3 to 20%. Levkoff and Sanchez [8] suggest minority recruitment and retention can be enhanced when there is a match between the goals of the ethnic minority community and the research community. The activity reported herein sought to enhance African American recruitment to a national cancer genetics research registry via collaboration between the National Educational Foundation (NEF) of the Zeta Phi Beta Sorority and the Mid-Atlantic Cancer Genetics Network (MACGN). This partnership sought to capitalize on the match between the NEF’s commitment to research and long-standing focus on genomic education, the Zeta Phi Beta Sorority’s interest in health and commitment to community outreach, and the MACGN’s experience in cancer genetics and research. This paper describes process, recruitment yield, and lessons learned from two recruitment strategies. Sharon J. Olsen, Assistant Professor Johns Hopkins University, School of Nursing 525 N Wolfe St, Rm 417 Baltimore, MD 21205 (USA) Tel. +1 301 972 0865, Fax +1 301 955 0466, E-Mail [email protected]

Zeta Phi Beta Sorority, Inc., and the NEF Zeta Phi Beta Sorority, Inc., is a national and international academic sorority. It was founded in 1920, on the campus of Howard University in Washington, D.C. Today, membership exceeds 100,000 college-educated and primarily African American women with a community service and education mission. Women’s health and breast and colon cancer prevention are important topics for community education. The NEF is the educational outreach arm of the Zeta Phi Beta Sorority. It was created and operates exclusively for charitable and educational purposes. Activities include awarding scholarships for higher education, conducting community education programs, and engaging in research. The NEF has a long-standing relationship with the Human Genome Project and the March of Dimes. In 1997, the NEF established a nationwide initiative, the Human Genome Project Information Conferences for Minority Communities. Five public education programs targeted minority citizens in New Orleans (1999), Philadelphia (2000), Atlanta (2001), Washington, D.C. (2001), and Chicago (2003). Topics included information about the Human Genome Project and an opportunity to discuss concerns about how genetic research might impact diverse ethnic and cultural groups. The conferences were advertised to the Zeta Phi Beta Sorority membership. Over 1,500 individuals participated in these conferences. Approximately 30 grass roots level conferences were subsequently supported by the NEF in minority churches, community centers and university settings across the United States. In 2002, the NEF compiled and published sixteen recommendations to share with decision-makers for use in planning genetics-related activities to meet the concerns and needs of all minorities. In July 2003, Sorority leaders met with legislators on Capitol Hill to discuss human genome research and minority concerns. The Mid-Atlantic Cancer Genetics Network The MACGN is one of eight academic centers of the Cancer Genetics Network (CGN) [9]. Established in 1998 and funded by the National Cancer Institute, the CGN makes it possible to do research that a single institution might not be able to do because of insufficient participant numbers or the time needed to recruitment them. The CGN supports collaborations to investigate the genetic basis of cancer susceptibility, explore mechanisms to integrate this new knowledge into medical practice, and identify ways to address associated psychosocial, ethical, legal, and public health issues. The MACGN recruitment 202

Community Genet 2008;11:201–207

area includes Maryland, Delaware, New Jersey, New York, southern Pennsylvania, northern Virginia and West Virginia. Regionally recruited participants are invited to enroll in the national CGN registry of prospective study participants who agree to serve as a potential participant pool for investigator-driven research on cancer genetic susceptibility, translational, and behavioral research. Enrollees complete a core questionnaire on family history and cancer risk factors. To enhance the CGN minority recruitment effort, the MACGN entered into a formal partnership with the NEF to design, implement and evaluate a pilot study to recruit African American women into the CGN registry. This paper describes the process and yield of two recruitment strategies across four states.

Methods Sample Two sampling frames constituted the convenience sample for this study. The first consisted of attendees at a 1-hour inherited breast cancer education and recruitment session presented during each of four State Leadership Conferences (Maryland, Pennsylvania, New Jersey and Delaware) (n = 279). The second consisted of 1,004 college-educated minority women listed as members of the Maryland, Pennsylvania, New Jersey and Delaware Zeta Phi Beta Chapters. These states were selected because of their proximity to MACGN. Eligibility criteria were consistent with MACGN and CGN recruitment eligibility and included: age 18 or older, self-described minority status, personal or family history of cancer, and ability to read and respond to an English language survey. Though little is known about the prevalence of hereditary cancer susceptibility in African American populations, the dominant perception is that at least 5–10% of all cancer is inherited – primarily in an autosomal dominant manner [10]. Extrapolating to this study, an expected eligibility of 64–128 subjects might be predicted. Study Design and Procedures This descriptive pilot study spanned October 2002 to September 2003 and was designed to fit within the existing regional Zeta Phi Beta Sorority conference structure. An important initial concern of the NEF leadership was appropriate human subjects review and approval. Analysis of the membership of the five Johns Hopkins University (JHU) Human Subjects Committees confirmed that at least one individual of minority/ethnic status was represented on each review board. Human subjects approval was obtained for the mutually agreed upon proposal. A memorandum of understanding was drafted by the immediate past Director of the NEF (a lawyer), reviewed by JHU council, and signed by the MACGN principal investigator and the NEF Director. As part of this agreement, each of four State Conferences received a USD 500 educational grant from the MACGN. The grant was awarded by the NEF. A mutually agreed upon promotional brochure was created to describe the collaboration and the nature of the recruitment effort.

Olsen /Malvern /May /Jenkins /Griffin

Entrée to the Directors of the four state Zeta Phi Beta chapters (Maryland, Pennsylvania, New Jersey, and Delaware) was provided by the NEF Director. The State Directors facilitated contacts with conference planning personnel. State Directors also checked in with MACGN personnel during the State Conferences. Recruitment employed two strategies, an onsite educational session and a two-tiered, personalized direct mail invitation. The four educational sessions occurred between March 2003 and May 2003. The targets were State Leadership Conferences (n = 4) held annually by the state Zeta Phi Beta chapters. These meetings are open and advertised to the local Zeta Phi Beta Sorority membership, friends of the Zeta Phi Beta known as members of the Amicae, and the greater community of minority women. MACGN personnel (a physician or advanced practice nurse) provided a 45to 60-min slide and discussion session at each meeting. Standardized content included: a brief overview of cancer genetics and what was known about inherited breast cancer in African Americans; a brief introduction to the CGN, MACGN, and the recruitment study; an interactive review of the registration packet, and an opportunity for questions and answers. A portfolio of materials was distributed to each attendee and included: a copy of the Powerpoint slides, the NEF/Zeta/MACGN promotional brochure, a MACGN brochure, four fact sheets (family history, inherited breast cancer, genetic testing, genetic counseling; all adapted for ethnic/racial sensitivity), a standardized packet of MACGN Registry Recruitment Materials (a standardized MACGN recruitment cover letter, a booklet describing the CGN, a core questionnaire, an informed consent form, a sample MACGN newsletter, a stamped, self-addressed envelope to return the questionnaire), and a promotional picture frame magnet and pen. All handouts were distributed in a 2-pocket portfolio designed for the study and featuring persons of color. Benefits to enrollment were discussed by study personnel and included access to the latest information about cancer genetics via a newsletter and referral to cancer genetics specialists throughout the United States for clinical services or advice as needed. Recruitment materials were coded to identify the conference location and to facilitate data analysis. Individuals could elect to (a) complete registry materials on site, (b) take the materials home and return the registration materials by mail in a stamped, selfaddressed envelope, or (c) call MACGN personnel using a tollfree number to request additional information or complete the registration by telephone. Contact information for the NEF was also included should conference attendees have any questions about the legitimacy of the research. A MACGN registry exhibit table was set up alongside other exhibitors to also promote the study. An interactive pedigree drawing touch screen program with a printout option was made available to promote the recording of personal family histories of cancer. The direct mail invitation involved two separate and staggered mailings to all members of each of the four state Zeta Phi Beta chapters. The first mailing (on NEF stationary) consisted of a signed cover letter of introduction and invitation from the NEF Director alerting Sorority members (Sorors) in each state to the new NEF/ZETA/MACGN Cancer Genetics Initiative study. A study brochure and MACGN brochure was included. The letter advised Sorors that they would receive a recruitment packet in the mail in 2 weeks from the MACGN at JHU. Each State Director

Table 1. African American recruitment response yields

Partnership

Community Genet 2008;11:201–207

Location

Approached

Undeliverable

Enrolled (response rate)

Presentation Maryland Pennsylvania New Jersey Delaware Subtotal

48 124 77 30 279 (22%)

NA NA NA NA

2 0 1 0 3 (1.075%)

Mailings Maryland Pennsylvania New Jersey Delaware Subtotal

371 314 275 44 1,004 (78%)

10 40 25 0 75 (7.5%)

11 6 6 1 24 (2.58%)

Total

1,283 (100%)

75

27 (2.23%)

facilitated access to their chapter’s membership and mailing list. MACGN personnel were responsible for collating and mailing all materials. The second mailing included a cover letter (on MACGN stationary) from both the NEF Chair and MACGN inviting interested Sorors to join the MACGN. This mailing included the standardized packet of MACGN registry recruitment materials and the four fact sheets. Those interested in joining the registry were given the choice to complete the core questionnaire by mail or telephone. If a Soror agreed to register by phone, an appointment was made to schedule an interview time. During the phone interview with a MACGN project coordinator, the participant was asked to have the consent form and questionnaire in hand. Verbal consent was obtained and then the questionnaire data were recorded. The participant was asked to return the signed written consent form by mail. Questionnaire data were not entered into the MACGN database until written consent was received. Unidentified MACGN data were transferred to the CGN at the University of California, Irvine. Statistical Considerations The on-site educational session yield was calculated based on the total number of signed consent forms and core data returned to the MACGN divided by the number of registration packets distributed during each educational session. The direct mail invitation recruitment yield was calculated based on the total number of signed consent forms and core data returned to the MACGN divided by the total number of mailings minus undeliverable addresses.

Results

Recruitment data through December 2004 are reported and summarized in table 1. In the onsite educational session recruitment mode, 279 participants attended the 203

targeted educational and recruitment presentations offered at the four State Leadership Conferences. Three individuals submitted core questionnaires, met the recruitment eligibility criteria and were enrolled in the CGN registry – resulting in a recruitment yield of 1.075%. The direct mail invitation was sent to 1,004 Sorors in four state chapters. Letters with forwarding addresses were forwarded and address lists updated for the second mailing. Seventy-five letters (7.5%) were undeliverable (74 bad addresses – primarily academic campus addresses; 1 death). This reduced the target sample to 929 Sorors who received the second mailing. Eight women contacted MACGN seeking further information or clarification. The recruitment yield for the direct mail invitation was 2.69% (n = 25). Overall, 28 women submitted applications. One self-described white female Sorority member was enrolled but is not included in summary data. The overall self-described African American recruitment yield was 2.23% (n = 27). Demographic characteristics are described in table 2. In general, women were married and well educated. The mean age at enrollment was 49. Two women had a personal history of breast cancer. Just over half of the sample identified a first-degree relative with cancer; about 78% listed a second-degree relative with cancer. Costs for printing, personnel, postage, travel, education and supplies for this study totaled USD 19,388. The average cost per successful African American enrollee was USD 718.

Discussion

To our knowledge, this is the first report of recruitment to a cancer genetics research registry targeting minorities associated with an academic Greek letter sorority. In this study, two separate recruitment efforts targeting healthy, well-educated African American members of a Greek sorority resulted in the recruitment of only 27 eligible minority women. The recruitment rate for this study falls short of the 3–20% rates previously published for minority participation in clinical research. The absolute number of those recruited also falls below the 5–10% of individuals initially projected to be eligible for participation due to a family history of cancer [10]. In a study of African Americans invited to participate in a national kidney disease and hypertension clinical trial, a mass mail campaign achieved a recruitment yield of only 1.1% [11]. In a cancer genetics trial by Royal et al. [12], successful recruitment 204

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Table 2. Characteristics of African American participants enrolled in registry (n = 27)

Females Age at enrollment, years 20–29 30–39 40–49 50–59 60–69 70–79 Mean SD Range Marital status Married or cohabiting Never married Divorced Widowed Separated Education College graduate or beyond Some college or technical school Ethnicity/racial background Black or African American No response Religion Protestant, NOS Baptist Christian, NOS Catholic Jewish Muslim 7th Day Adventist No response Personal history of breast cancer Family history of cancer First-degree relatives Second-degree relatives Third-degree relatives First- and second-degree relatives First-, second- and third-degree relatives

n

%

27

100

3 7 7 1 5 4 48.9 16.55 29–77

11.1 25.9 25.9 3.7 18.5 14.8

12 6 4 4 1

44.4 22.2 14.8 14.8 3.7

25 2

92.5 7.4

26 1

96.3 3.7

13 4 4 2 1 1 1 1 2

48.1 14.8 14.8 7.4 3.7 3.7 3.7 3.7 7.4

14 21 6 11 3

51.8 77.7 22.2 40.1 11.1

NOS = Not otherwise specified.

of African American men into a hereditary prostate cancer study was best enhanced by physician referral and the use of tumor registries. Researchers with the CarolinaGeorgia CGN examined racial differences in recruitment to their CGN registry [13]. They had targeted women previously enrolled in a breast cancer research study and found lower rates of recruitment among African American women with a personal or family history of breast cancer (15%) compared with similar white women (36%) Olsen /Malvern /May /Jenkins /Griffin

[13]. Enrollment was not explained by socioeconomic factors, though there was an enrollment trend toward younger women with higher incomes. The authors speculated that differences in perceptions about the purpose, benefit, burden and/or risk of involvement in the registry compared to participation in the previous research study may have raised more concern among African American women. A possible reason for the low accrual to the current study might be that women were asked to enroll in a research study wherein no immediate benefit for joining was apparent and where they may or may not be invited to participate in future cancer genetics studies depending on eligibility. Barriers to African American participation in cancer genetics research have been known to include concerns about exploitation [14, 15], greater levels of psychological distress [16], reduced knowledge about or exposure to information about cancer genetics or inherited cancer [16– 19] and discrimination, stigmatization and confidentiality [17]. Historically, issues concerning stigmatization, discrimination, privacy and confidentiality have been openly discussed in national NEF sponsored genome conferences. Concerns about confidentiality were discussed at each educational session. Privacy and confidentiality were recurring concerns voiced in the question and answer periods at the onsite educational sessions. Questions included: ‘How will my family history information be protected? What confidentiality protections had been implemented? What will happen with the data from the questionnaires that I complete for this recruitment study? How is personal information protected in the national database?’ A number of limitations bear consideration. First, it is possible that a small number of Sorority members may have been exposed to more than one recruitment mode. Members attending any one of the four state leadership meetings might have also received a targeted mailing as the educational session attendance list was not available for comparison with the membership roster. Second, recruitment to research studies is not common at most Sorority events and awareness of this study was generally limited to those attending the educational session on inherited breast cancer. Due to time constraints, no preconference advertisement was distributed. The only advanced notification about the study was limited to inclusion of the NEF/Zeta/MACGN study brochure and a one-page flyer about the presenters in the conference packet which was distributed at the time of conference registration. Clearly, preconference announcements or advertisement in program brochures may have better

prepared attendees. Third, the educational session presenters were generally white women representing a lack of concordance between the recruiters and potential participants. Moorman et al. [2] suggest this may negatively influence minority recruitment. However, discussion with the NEF Director and conference planners concluded this was likely not a significant issue in this study as it was common to have ethic/culturally diverse speakers at Zeta Phi Beta conferences. The MACGN personnel were always introduced by the Zeta Phi Beta regional directors or conference personnel. They were also acknowledged for their participation at the conference luncheons. It is noteworthy that on the one occasion that study personnel included a minority representative from the University of Pennsylvania, no increase in recruitment was noted. Lastly, data were not collected that could further our understanding of whether recruitment materials were shared with family or friends, how Sorors perceived this information, or why eligible Sorors chose not to participate.

Partnership

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Lessons Learned Data from this study were presented to the Zeta Phi Beta Sorority leadership. They too expressed concern about the low recruitment numbers. However, there was clear interest in extending this study nationally and working together to attain funding for a larger study. Second, some Sorority members commented that they did not open the introductory mailing from the NEF Chair, so when they got the MACGN mailing, there was concern that MACGN had inappropriate access to their address. To avoid this in the future, it would be useful to tailor the face of all mailed materials in a way that can be differentiated from the usual NEF mailings. Third, open discussions with conference participants acknowledged that African American families tend not to share personal or family medical histories within or across generations. In fact, attendees clearly acknowledged a tendency toward secrecy around family illnesses and diseases. Conference attendees articulated the need to initiate family discussions about inheritance in order to protect the health of their loved ones. Because extended family reunions are common among many African American families, such gatherings should be recognized as a strategic opportunity to tap into the hidden knowledge and experience of elders and to begin to develop and share family health histories. Additionally, because there was a preference for interactive educational sessions, future conference activities could include opportunities to build skills in recording a 3-generation family history. 205

Fourth, despite the apparent match between the goals of the NEF, the Zeta Phi Beta Sorority, and the MACGN, minority recruitment was not enhanced and the findings of this study do not support the work of Levkoff and Sanchez [8]. However, an important strength of working with traditional Greek letter organizations such as the Zeta Phi Beta Sorority is their focus on community service and outreach. Each Zeta Phi Beta Sorority member is expected to personally reach out to their local community to share health information. Fifth, because the Zeta Phi Beta Sorority is committed to community outreach, the leadership supports the development of a standardized education training manual for its members. This manual provides specific information about how to set up a community workshop or short presentation, what information to deliver (including resources for ordering brochures), and how to evaluate each session. Manuals could include modules on genetics, inherited cancer risks and pedigree development. Sixth, it is important to recognize that volunteer organizations have many competing interests. This necessitates investigator-initiated contact and communication hypervigilance. The major players in this study were geographically dispersed. E-mail and Internet access were critical to rapid review and turn around of the proposal, memorandum of understanding, brochure text, and selection of graphic art. The MACGN and JHU infrastructure provided support for brochure design, xeroxing, mailing and envelope stuffing. Technical support from MACGN helped convert membership lists to formats that facilitated personalization of cover letter and mailings. Lists were updated based on mailing responses and returned to each state office. Flexibility to respond quickly to requests for summary information at follow-up meetings of the Zeta Phi Beta leadership to report findings was also important.

Finally, the NEF and state chapters went out of their way to integrate the educational and recruitment activities for this study into already developed and full conference schedules. Advance planning for a session immediately before or after the planned state conference or a dinner session might facilitate recruitment, as might appearances at local meetings. These were not possible given the time frame available for this study. In summary, this study examined two sorority-based strategies for recruiting minority participants to a national Cancer Genetics Registry for the purpose of participation in research studies about cancer genetic susceptibility and translational or behavioral research. Recruitment efforts yielded a very modest 27 eligible and enrolled African American women. Extending recruitment to the national Zeta Phi Beta Sorority was recommended and encouraged by the Zeta Phi Beta Sorority leadership. This study adds to a growing body of literature concerning minority recruitment to cancer genetics research. In the future, collaboration with minority sororities and fraternities could become part of standard procedures for registry recruitment.

Acknowledgements The authors wish to acknowledge and thank the following for their support in this study: Jacqueline Lemon-Denton, Atlantic Regional Director; Linda J. Amos, Delaware State Director; Janet Y. Bivins, Pennsylvania State Director; Pauline E. Gibson, New Jersey State Director; Jacqueline McNair, Maryland State Director; the membership of Zeta Phi Beta Sorority, Inc., in each of these four states and Stacey Armstrong, Research Assistant for her support with the two-tiered direct mail campaign and data collection. This study was funded by 5U24CA78148.

References 1 National Institutes of Health: NIH guidelines on the inclusion of women and minorities as subjects in clinical research. Fed Regist 1994;59:14508–14513. 2 Moorman PG, Newman B, Millikan RC, Tse CK, Sandler DP: Participation rates in a casecontrol study: the impact of age, race, and race of interviewer. Ann Epidemiol 1999; 9: 188–195. 3 Richardson MA, Post-White J, Singletary SE, Justice B: Recruitment for complementary/ alternative medicine trials: who participates after breast cancer. Ann Behav Med 1998;20: 190–198.

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4 Lerman C, Hughes C, Benkendorf JL, Biesecker B, Kerner J, Willison J, Eads N, Hadley D, Lynch J: Racial differences in testing motivation and psychological distress following pretest education for BRCA1 gene testing. Cancer Epidemiol Biomarkers Prev 1999; 8: 361–367. 5 Moinpour CM, Atkinson JO, Thomas SM, Underwood SM, Harvey C, Parzuchowski J, Lovato LC, Ryan AM, Hill MS, Deantoni E, Gritz ER, Thompson IM Jr, Coltman CA Jr: Minority recruitment in the prostate cancer prevention trial. Ann Epidemiol 2000; 10: S85–S91.

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6 Gifford AL, Cunningham WE, Heslin KC, Andersen RM, Nakazono T, Lieu DK, Shapiro MF, Bozzette SA: HIV Cost and Services Utilization Study Consortium: participation in research and access to experimental treatments by HIV infected patients. N Engl J Med 2002;346:1373–1382. 7 Adams-Campbell LL, Ahaghotu C, Gaskins M, Dawkins FW, Smoot D, Polk OD, Gooding R, DeWitty RL: Enrollment of AAs into clinical treatment trials: study design barriers. J Clin Oncol 2004;22:730–734.

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8 Levkoff S, Sanchez H: Lessons learned about minority recruitment and retention from Centers on Minority Aging and Health Promotion. Gerontologist 2003; 43:18–26. 9 Anton-Culver H, Ziogas A, Bowen D, Finkelstein D, Griffin C, Hanson J, Isaacs C, Kasten-Sportes C, Mineau G, Nadkarni P, Rimer B, Schildkraut J, Strong L, Weber B, Winn D, Hiatt R, Nayfield S: The Cancer Genetics Network: Recruitment results and pilot studies. Community Genet 2003;6:171–177. 10 Nagy R, Sweet K, Eng C: Highly penetrant hereditary cancer syndromes. Oncogene 2004;23:6445–6470. 11 Whelton PK, Lee JY, Kusek JW, Charleston J, DeBruge J, Douglas M, Faulkner M, Greene PG, Jones CA, Kiefer S, Kirk KA, Levell B, Norris K, Powers SN, Retta TM, Smith DE, Ward H: Recruitment experience in the African American study of kidney disease and hypertension (AASK) pilot study. Control Clin Trials 1996;17:17s–33s.

Partnership

12 Royal C, Baffoe-Bonnie A, Kittles R, Powell I, Bennett J, Hoke G, Pettaway C, Weinrich S, Vijayakumar S, Ahaghotu C, Mason T, Johnson E, Obeikwe M, Simpson C, Mejia R, Boykin W, Roberson P, Frost J, Faison-Smith L, Meegan C, Foster N, Furbert-Harris P, Carpten J, Bailey-Wilson J, Trent J, Berg K, Dunston G, Collins F: Recruitment experience in the first phase of the African American Hereditary Prostate Cancer (AAHPC) Study. Ann Epidemiol 2000; 10:S68–S77. 13 Moorman PG, Skinner CS, Evans JP, Newman B, Sorenson JR, Calingaert B, Susswein L, Crankshaw TS, Hoyo C, Schildkraut JM: Racial differences in enrolment in a cancer genetics registry. Cancer Epidemiol Biomarkers Prev 2004;13:1349–1354. 14 Hughes C, Gomez-Caminero A, Benkendorf J, Kerner J, Isaacs C, Barter J, Lerman C: Ethnic differences in knowledge and attitudes about BRCA1 testing in women at increased risk. Patient Educ Counsel 1997;32:51–62.

15 Lerman D, Audrain J, Croyle R: DNA-testing for heritable breast cancer risks: lessons from traditional genetic counseling. Ann Behav Med 1994:16:327–333. 16 Halbert CH, Kessler L, Collier A, Paul Wileyto E, Breswter K, Weathers B: Psychological functioning in African American women at an increased risk of hereditary breast and ovarian cancer. Clin Genet 2005; 68: 222– 227. 17 Donovan KA, Tucker DC: Knowledge about genetic risk for breast cancer and perceptions of genetic testing in a sociodemographically diverse sample. J Behav Med 2000; 23: 15–36. 18 Peters N, Rose A, Armstrong K: The association between race and attitudes about predictive genetic testing. Cancer Epidemiol Biomarkers Prev 2004; 13:361–365. 19 Wideroff L, Vadaparampil ST, Breen N, Croyle RT, Freedman AN: Awareness of genetic testing for increased cancer risk in the year 2000 National Health Interview Survey. Community Genet 2003;6:147–156.

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Community Genet 2008;11:208–214 DOI: 10.1159/000116881

Successful Strategies for Increasing African American Participation in Cancer Genetic Studies: Hopeful Signs for Equalizing the Benefits of Genetic Medicine Annette R. Patterson a Helen Davis a Kristin Shelby a Jerry McCoy a Linda D. Robinson a Smita K. Rao a Pia Banerji a Gail E. Tomlinson a, b a Simmons Comprehensive Cancer Center and b Departments of Pediatrics and Internal Medicine, University of Texas Southwestern Medical Center, Dallas, Tex., USA

Key Words Minority recruitment ⴢ Hereditary breast cancer ⴢ African American ⴢ Cancer genetic studies

Abstract Objective: To determine whether prior success in recruiting African Americans to an in-house cancer genetics registry could be duplicated when recruiting to a national registry requiring a significantly increased level of commitment. Additionally, to determine which recruitment sources and practices yielded the highest number of African American participants. Methods: A retrospective analysis of recruitment sources, practices, and results for recruitment to the Cancer Genetics Network (CGN; a national research registry), from 2000 to 2005 was conducted. These results were compared to previous experience in recruiting African Americans to the Family Cancer Registry (FCR; an in-house registry) during the period 1992–2005. Results: In the 1st year of recruitment to the CGN, African Americans accounted for 24% of those consenting to participate in the CGN registry from our center. This compares to an average annual rate of 27% for the FCR during the years 1998–2005, and a rate of less than 1% from 1992 to 1998. By 2005, African Americans accounted for 27% of CGN participants recruited through the University of Tex-

© 2008 S. Karger AG, Basel 1422–2795/08/0114–0208$24.50/0 Fax +41 61 306 12 34 E-Mail [email protected] www.karger.com

Accessible online at: www.karger.com/cmg

as Southwestern Medical Center, one of eighteen participating institutions in the CGN. Hospital-based resources such as cancer treatment clinics and tumor registries yielded the highest percentage of African American participants (66.5%), and self-referral yielded the lowest (0%). Seventy-seven percent of African Americans were actively sought out and recruited from treatment clinics, whereas the vast majority of Caucasian participants were recruited passively during the course of genetic counseling sessions that were scheduled for reasons unrelated to participation in cancer research. There were no known instances of African Americans contacting CGN staff after reading printed recruitment materials or internet advertisements. Conclusions: The increased level of commitment required of CGN participants did not deter African Americans from participating in cancer genetics research. Recruitment strategies responsible for dramatically increasing recruitment rates to the FCR from 1998 to 2000 were equally effective when used for recruitment to the CGN. The most effective recruitment sources were highyield venues such as cancer treatment clinics and tumor registries, and active recruitment methods yielded the highest number of African American participants. Advertising through internet announcements and printed recruitment materials did not appear to be effective. Copyright © 2008 S. Karger AG, Basel

Gail Tomlinson, MD, PhD University of Texas Health Science Center of San Antonio, Department of Pediatrics 7703 Floyd Curl Drive San Antonio, TX 78229 (USA) Tel. +1 210 704 3111, Fax +1 210 704 3577, E-Mail [email protected]

Introduction

The Clinical Cancer Genetics unit at the University of Texas Southwestern Medical Center is part of a network of clinics and affiliated hospitals providing medical care in the Dallas-Fort Worth metroplex. Serving patients funded through private insurance companies as well as public institutions, our recruitment base for cancer genetic studies is both ethnically diverse and highly varied with regard to educational background and socioeconomic status. The majority of African American research participants are recruited through Parkland Hospital, a county facility serving primarily low-income families. As of 2005, 20.9% of individuals in the catchment area for this facility identified as African American [1]. Barriers to participation in this population are both numerous and complex, though our experience and the experience of others [2–5] suggest that such barriers can be overcome through the expenditure of additional time and resources and through strategic initiatives designed to increase participation of African Americans. An important concept in recruiting underserved populations is that of active versus passive recruitment methods. As described by Lee et al. [6] in 1997, active recruitment methods are those requiring additional expenditure of energy and resources in order to identify appropriate research candidates and facilitate participation. Examples of active methods used at our center include use of tumor registries to identify high-risk patients, maintaining an ongoing presence in cancer clinics, and meeting with individuals on evenings and weekends in order to circumvent scheduling difficulties. Passive recruitment methods require little or no expenditure of additional resources and include methods such as simply providing information about the study following referral from a physician or recruiting participants who self-identify as high risk and self-refer for participation in research studies. From 1992 to 1998 investigators from our team had very limited success in recruiting African Americans for cancer genetic studies (recruitment rate less than 1%), though we were generally successful in recruiting other populations. Suspecting that issues of trust and a lack of identifying with our team might be part of the problem, we hired an African American nurse with 18 years of experience in patient care to coordinate recruitment efforts. In addition to being an active member of the African American community, she is also affiliated with a number of cancer support groups and organizations that provide education on cancer-related topics. She regularly attends cancer treatment and high-risk clinics, which enIncreasing African American Participation in Cancer Genetic Studies

ables her to become acquainted with cancer patients and family members before discussing the possibility of participation in cancer genetics research. Repeat exposure over an extended period of time also appears to have been important in a number of cases in which participants enrolled only after interacting with the recruiter on multiple occasions and being offered participation a number of times. In 1998, the initial year after her hiring, our recruitment rate for African Americans exceeded that of the previous 6 years combined. In 2000, as part of a national network of institutions participating in the Cancer Genetics Network (CGN), we began recruiting individuals to the CGN registry. The primary goal of this network was to establish a national registry of high-risk individuals willing to be contacted for repeat participation in cancer genetic studies. This registry was intended for use by ‘the research community at large to support studies on the genetic basis of human cancer susceptibility’ [7]. A central database was created to house detailed information on participants that would facilitate research on ‘both common and uncommon tumors’ [7]. Given the increased level of commitment required of CGN participants (as opposed to our own local registry, which requires only a one-time blood draw and completion of a registry questionnaire), the question of whether previous recruitment success could be duplicated was an interesting one.

Methods Recruitment Sources The recruitment source for each patient was determined using data collected by the research coordinator. Recruitment sources included hospital-based cancer care clinics (both public and private), tumor registries, and social and religious networks providing cancer-related education and support. Recruitment Strategies From 2000 to 2005, African Americans were recruited to the CGN registry using the same strategies first implemented by our group in 1998 in attempting to increase participation of African Americans in our Family Cancer Registry (FCR). Promotion of CGN initiatives and identification of potential participants were accomplished primarily through personalized (face-to-face) interactions in cancer clinics and cancer-specific community outreach activities. A number of African American participants were enrolled at remote locations during evenings and weekends in an attempt to reduce barriers to participation, both real and perceived. In two instances, recruitment was coupled with all-day educational seminars designed to meet the needs of specific hereditary cancer families. During these educational sessions, research staff addressed a broad range of topics geared toward increasing knowledge of basic genetic principles, familiarizing

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209

700

FCR

600

CGN

500 400 300 200 100 0

1992

1994

1996

1998

2000

2002

2004

Fig. 1. Cumulative recruitment of African Americans to the FCR

(1992–2005) and the CGN (2000–2004) at UT Southwestern Medical Center.

family members with risk reduction strategies, and informing families of opportunities to participate in research. In one other instance, research staff attended the family reunion of a known hereditary cancer family, enrolling and counseling multiple branches of this family throughout the day. In addition to the strategies outlined above, targeted recruitment materials were placed in clinic waiting rooms and physicians’ offices including the ‘Buena Vida Guide to Family Cancer Research’ [8], a bilingual booklet of basic concepts in genetics that offered participation in genetics studies, and a one-page instructional flyer entitled, ‘What’s in Your Family Tree?’ produced by genetics personnel at UT Southwestern Medical Center, through a previous grant from the local affiliate of the Susan G. Komen Breast Cancer Foundation [9]. The CGN also advertised through a website describing the purpose and goals of the CGN, which also listed the contact information of participating institutions [7]. Strategies including use of other mass media and large-scale mailings were not employed by our group.

Results

Recruitment In the first year of recruitment, African Americans accounted for 24% (16/66) of those consenting to participate in the CGN registry (compared to an average annual rate of 27% for the FCR during the years 1998–2005). By 2005, African Americans accounted for 27% (233/ 875) of all CGN participants recruited at our site. From 1998 to the present, African Americans accounted for 27% (532/1992) of those enrolled in the FCR. Figure 1 shows cumulative recruitment for both the FCR (1992– 2005) and the CGN (2000–2005). 210

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Recruitment Sources The majority of African American participants (66.5% or 155/233) were recruited to the CGN through hospitalbased resources such as cancer treatment clinics and tumor registries. Of these, 66% (102/155) were actively recruited through the county medical facility serving mostly low-income patients and 34% (53/155) were offered participation during a genetic counseling session following referral from private clinics (passive recruitment). Expansion of families originally ascertained through hospital-based resources accounted for 28% (65/233) of individuals recruited to the CGN, though recruitment for this group took place primarily in remote locations such as churches and recreation facilities where large family gatherings were held. Home visits were also instrumental in recruiting those unable or unwilling to participate in family gatherings. A small number (5.5% or 13/233) were recruited through social and religious networks providing cancer-related education and support. 91% of African Americans recruited to the CGN were female, reflecting recruiter focus on breast cancer treatment clinics. Recruitment Strategies Effective methods for recruitment of African American participants differed markedly from those for Caucasians. Whereas the majority of Caucasians (190%) were recruited passively during a genetic counseling session scheduled for other reasons, the majority of African Americans (77% or 180/233) were actively recruited while waiting for appointments in cancer treatment clinics, at family gatherings attended by research staff, or through social and religious networks providing education and support. Thus, recruitment methods for African Americans were primarily active and for Caucasians, primarily passive. In addition, though a significant number of Caucasian individuals self-referred for participation after reading printed materials and/or internet advertisements, not a single African American self-referred for participation. There were no known instances of African Americans contacting research staff after reading study materials. Recruitment Rate Offers of participation in the CGN were not consistently recorded, precluding provision of a specific recruitment rate. Based on available data and recruiter assessment, we estimate that 190% of African Americans who were offered participation in the CGN accepted.

Patterson /Davis /Shelby /McCoy / Robinson /Rao /Banerji /Tomlinson

Discussion

Over the last decade, the discovery of high-penetrance cancer predisposition genes has revolutionized the management and treatment of hereditary cancer families. Rapid development of genetic counseling and testing protocols has made it possible to identify high-risk individuals before cancer occurs, allowing for implementation of early detection and prevention strategies. Though subsequent research has proven the efficacy of using genetic information to estimate and reduce the risk of some cancers [10–13], the medical literature also indicates that not all populations and ethnicities enjoy equal benefit from these advances [14]. Notably, in the United States, African Americans currently account for only 3% of individuals who have undergone analysis of the BRCA1 and BRCA2 cancer predisposition genes [15] and only 3.2% of those tested by Myriad Genetic Laboratories for Hereditary Non-Polyposis Colon Cancer [Myriad Genetic Laboratories, pers. commun.]. Recent studies suggest that fear of discrimination and stigmatization may provide a partial explanation for these findings [2, 16–20], in addition to factors such as lack of knowledge regarding cancer genetics [18–19]. These trends are particularly worrisome, given that black Americans have the highest incidence of cancer overall, and are 33% more likely to die of cancer than their white counterparts [21]. In addition to limited use of genetic services, the medical literature suggests that African Americans are less likely to participate in cancer genetics research [3, 22] and to take advantage of research protocols offering genetic counseling and testing [17, 18, 23–26]. In one such study, ⬃22% of African American women declined the opportunity to undergo genetic counseling and 25% underwent genetic counseling but declined genetic testing, in spite of the fact that women in both of these groups strongly endorsed genetic testing [17]. In an ongoing study at our own institution entitled ‘Decision Making and BRCA Testing in African American Women’, 15% of women contacted thus far (4/26) have declined genetic counseling and 15% of those counseled (2/13) have declined genetic testing. Reasons given for the decision not to test include fear of the consequences of genetic testing for family members, a finding that is consistent with results from previous studies [17, 18, 25–27]. This reluctance to participate in genetic research and testing on the part of African Americans is of critical importance, as it undermines the establishment of a sound informational basis on which development of population-specific genetic counseling and testing protocols depends. It also restricts Increasing African American Participation in Cancer Genetic Studies

the potential benefit of genetic medicine, one of the more promising new avenues for reducing cancer risk and decreasing cancer disparities between Caucasian and minority populations. These various findings establish an urgent need for finding ways to increase African American participation in cancer genetics research. To address these and other recruitment issues, we have spent the last 9 years developing a research infrastructure aimed at increasing minority participation in cancer genetics research. During this period, we employed a number of strategies with varied success. Our current strategy reflects lessons learned over time and focuses primarily on increasing comfort level and reducing the ‘burden’ of research participation. Both become critically important when attempting to recruit a population that is more likely to receive care at county facilities for low-income patients and less likely to self-refer for genetic services. Key elements of our recruitment strategy involve using cost-effective methods for identification of high-risk African Americans and addressing known barriers to participation in cancer genetics research. Recruiting from high-risk clinics and cancer treatment centers restricts the potential recruitment pool to individuals with a personal history of cancer or a specific risk profile. Employing an African American nurse to coordinate recruitment efforts and maintain a clinic presence allows us to increase visibility among patients and physicians, while addressing barriers to participation such as lack of trust as well as time or transportation issues. Cancer-specific outreach efforts through cancer support groups, educational forums, and church-sponsored events provide additional opportunities for recruitment. Flexibility with regard to scheduling is also critically important, as many of our African American participants report having inflexible work schedules and difficult extenuating circumstances. For these individuals, participation in cancer genetic research often hinges on reducing logistical burden. By coupling CGN recruitment with regularly scheduled appointments we were able to enroll patients without asking for additional personal time or requiring that they make additional trips to the clinic. Home visits during evenings and weekends allowed us to address other barriers such as transportation difficulties, time constraints, and psychological resistance to the clinic setting. Holding weekend family gatherings in churches and recreation facilities provided an atmosphere that was comfortable and familiar and allowed us to reach large numbers of family members who might not participate otherwise due to scheduling and transportation issues. These Community Genet 2008;11:208–214

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gatherings were often scheduled at the request of individuals who had previously enrolled in studies and were extremely concerned about family cancer risk. Though held primarily for educational purposes, information about opportunities for research participation was routinely included. Individuals wishing to participate signed consent forms approved by the IRB at the University of Texas Southwestern Medical Center. Such gatherings also allowed us to increase productivity and to use time and resources more efficiently. Based on our own observations and the observations of others [3–5, 28, 29], flexibility with regard to scheduling and emphasis on issues of trust are two key aspects of a successful recruitment strategy for African Americans. A number of studies have now shown that personalized recruitment strategies are effective for enrolling African Americans for cancer genetic studies [3, 19, 30]. One possible explanation for this is that personalized interactions allow the recruiter to anticipate and address potential barriers to participation such as fear of discrimination and mistrust of the medical establishment before such barriers form the basis of a negative response. Potential participants are also afforded the opportunity to ask questions and discuss their fears about participating in cancer genetics research [4]. Perhaps most importantly, such interactions provide an opportunity to introduce basic genetic principles and to familiarize African Americans with recent advances in both our ability to pinpoint the underlying causes of hereditary disease and our ability to reduce cancer risk through genetic testing and treatment protocols. Studies have also shown that the benefit of personalized interaction can be further enhanced through concordance [3, 31]. The dramatic increase we experienced after hiring an African American nurse supports these findings, though it is difficult to separate concordance from other factors that may have influenced recruitment rate, such as maintaining an ongoing presence in the clinic. To the extent that concordance was responsible for altering recruitment rates, the most likely interpretation is that it effectively countered well-documented mistrust of the medical establishment [2, 5, 18, 20, 28, 29, 32, 33] among African Americans. In order to retain African American participants, we developed a number of strategies that include coordinating and sponsoring educational events in the African American community, contacting participants by phone to update family cancer histories, interacting with participants in cancer clinics as they return for treatment or ongoing management, and sending annual letters to re212

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search participants during the December holidays. As it is customary in our society during this time of year to make a special effort to connect with others, to wish them well, to reflect on the current year and to anticipate the promise of the year to come, our practice of sending holiday letters allows us to extend goodwill to participants, to update participants on current studies, and to provide information regarding additional opportunities for research participation. At the close of our letter, we ask that participants actively respond by writing, e-mailing, or calling the recruitment coordinator to provide follow-up information regarding personal well-being and any significant changes in cancer family history. Limitations of the Study The limitations of this study include the fact that it is an historical comparison and not a prospective randomized controlled study comparing intervention to nonintervention groups. This makes it difficult to draw firm conclusions, particularly with regard to recruitment sources and practices. In addition, though the recruiter reports that African American individuals who were offered participation during the period 1998–2005 rarely declined, failure to record all offers and outcomes precludes provision of exact recruitment rates. Though this study provides preliminary insight into various factors affecting African American participation in cancer genetic studies, prospective, randomized studies are needed to confirm and expand upon these findings.

Conclusions

Increasing participation of minorities in cancer genetics research is critically important. This is particularly true for African Americans, who are disproportionately affected by cancer burden, but currently underrepresented among those seeking genetic services and those participating in cancer genetic studies. In this study we explored possible barriers to participation and evaluated various strategies designed to increase participation, particularly among low-income African American women. We found that the increased level of commitment required of CGN participants did not deter African Americans from participating in cancer genetics research. Recruitment strategies that were responsible for dramatically increasing participation in our local registry (FCR) from 1998 to 2000 were equally effective when used for recruitment to the CGN. The most effective recruitment sources were high-yield venues such as cancer treatment Patterson /Davis /Shelby /McCoy / Robinson /Rao /Banerji /Tomlinson

clinics and tumor registries, and active recruitment methods yielded the highest number of African American participants. Advertising through internet announcements and printed recruitment materials did not appear to be effective. As the field of medical genetics moves forward and the promise of genetic testing is realized, we must take care not to replicate the inequalities of the past. Ensuring African Americans a benefit equal to that of Caucasians will require aggressive efforts to enlist African Americans in the investigative process. Successful development of population-specific protocols will depend on our ability to recruit African Americans for cancer genetic studies and to maintain an active research base in the African American population. Sustained progress in clarification of penetrance rates, mutation spectrum, and cultural fac-

tors influencing use of genetic testing will inevitably lead to development of population-specific protocols. It is our hope that this will increase both the efficacy of genetic counseling/testing in reducing cancer risk and the economic feasibility of genetic testing through identification of recurrent mutations in the African American population.

Acknowledgements Targeted initiatives described in this paper were almost entirely supported through grant funding from private foundations and federal agencies. Grants subsidizing these efforts include: NIH R03-CA70472 (GT), Susan G. Komen Breast Cancer Foundation Grant No. POP0201126 (GT) and NIH U24-CA78142 (Texas Cancer Genetics Consortium, part of the CGN).

References 1 US Census Bureau: Quick Facts from the US Census Bureau. http://quickfacts.census.gov/qfd/states/48/48113/html, accessed 6/20/2007. 2 Royal C, Baffoe-Bonnie A, Kittles R, Powell I, Bennett J, Hoke G, Pettaway MD, Weinrich S, Vijayakumar S, Ahaghotu C, Mason T, Johnson E, Obeikwe M, Simpson C, Jejia R, Boykin W, Roberson P, Frost J, Faison-Smith L, Meegan C, Foster N, Furbert-Harris P, Carpten J, Bailey-Wilson J, Trent J, Berg K, Dunston G, Collins F: Recruitment experience in the first phase of the African American Hereditary Prostate Cancer (AAHPC) Study. Ann Epidemiol 2000; 10:S68–S77. 3 Hughes C, Peterson SK, Ramirez A, Gallion KJ, McDonald PG, Skinner CS, Bowen D: Minority recruitment in hereditary breast cancer research. Cancer Epidemiol Biomarkers Prev 2004;13:1146–1155. 4 Patterson AR, Davis H, Euhus D, Neuhausen S, Strong L, Tomlinson GE: Recruitment for breast cancer predisposition studies in an underserved African American population. Breast J 2005; 11:79–82. 5 McCaskill-Stevens W, McKinney MM, Whitman CG, Minasian LM: Increasing minority participation in cancer clinical trials: The Minority-Based Community Clinical Oncology Program experience. J Clin Oncol 2005;23:5247–5254. 6 Lee RE, McGinnis KA, Sallis JF, Castro CM, Chen AH, Hickmann SA: Active vs. passive methods of recruiting ethnic minority women to a health promotion program. Ann Behav Med 1997;19:378–384.

Increasing African American Participation in Cancer Genetic Studies

7 National Cancer Institute website for Cancer Control and Population Sciences: Epidemiology and Genetics Research; Cancer Genetics Network. http://epi.grants.cancer.gov/ CGN/, accessed 9/11/2005. 8 Texas Cancer Genetics Consortium: Buena Vida: A Guide to Family Cancer Research. http://texas.cgnweb.org. 9 Steiglitz H: What’s in Your Family Tree? San Antonio, University of Texas Southwestern Medical Center, 1995. 10 Syngal S, Weeks JC, Schrag D, Garber JE, Kuntz KM: Benefits of colonoscopic surveillance and prophylactic colectomy in patients with hereditary nonpolyposis colorectal cancer mutations. Ann Intern Med 1998;29: 787–796. 11 Rebbeck TR, Levin AM, Eisen A, Snyder C, Watson P, Cannon-Albright L, Isaacs C, Olopade O, Barber JE, Godwin AK, Daly MB, Narod SA, Neuhausen SL, Lynch HT, Weber BL: Breast cancer risk after bilateral prophylactic oophorectomy in BRCA1 mutation carriers. J Natl Cancer Inst 1999; 91: 1442– 1443. 12 Kauff ND, Satagopan JM, Robson ME, Scheuer L, Hemsley M, Hudis CA, Ellis NA, Boyd J, Borgen PI, Barakat RR, Norton L, Castiel M, Nafa K, Offit K: Risk-reducing salpingo-oophorectomy in women with a BRCA1 or BRCA2 mutation. N Engl J Med 2002;346:1609–1615. 13 Rebbeck TR, Lynch HT, Neuhausen SL, Narod SA, Van’t Veer L, Garber JE, Evans G, Issacs C, Daly MB, Matloff E, Olopade OI, Weber BL, Prevention and Observation of Surgical End Points Study Group: prophylactic ophorectomy in carriers of BRCA1 or BRCA2 mutations. N Engl J Med 2002; 346: 1616–1622.

14 Olopade OI: Genetics in clinical cancer care: a promise unfulfilled among minority populations. Can Epidemiol Biomarkers Prev 2004;13:1683–1686. 15 Pal T, Permuth-Wey J, Holtje T, Sutphen R: BRCA1 and BRCA2 mutations in a study of African American breast cancer patients. Can Epidemiol Biomarkers Prev 2004; 13: 1794–1799. 16 Peterson EA, Milliron KJ, Lewis KE, Goold SD, Merajver SD: Health insurance and discrimination concerns and BRCA1/2 testing in a clinic population. Cancer Epidemiol Biomarkers Prev 2002; 11:79–87. 17 Thompson HS, Valdimarsdottir HB, Dutear-Buck C, Guevarra J, Bovbjerg DH, Richmond-Avellaneda R, Amarel D, Godfrey D, Brown K, Offit K: Psychosocial predictors of BRCA counseling and testing decisions among urban African-American women. Cancer Epidemiol Biomarkers Prev 2002;11: 1579–1585. 18 Hughes C, Gomez-Caminero A, Benkendorf J, Kerner J, Issacs C, Barter J, Lerman C: Ethnic differences in knowledge and attitudes about BRCA1 testing in women at increased risk. Patient Educ Couns 1997; 32:51–62. 19 Donovan KA, Tucker DC: Knowledge about genetic risk for breast cancer and perceptions of genetic testing in a sociodemographically diverse sample. J Behav Med 2000; 23: 15–35. 20 Phillips JM, Cohen MZ, Moses G: Breast cancer screening and African American women: fear, fatalism, and silence. Oncol Nurs Forum 1999;26:561–571. 21 Cancer Facts and Figures 2001. Atlanta, American Cancer Society, 2001.

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22 Moorman PG, Skinner CS, Evans JP, Newman B, Sorenson JR, Calingaert B, Susswein L, Crankshaw TS, Hoyo C, Schildkraut JM: Racial differences in enrolment in a cancer genetics registry. Cancer Epidemiol Biomarkers Prev 2004;13:1349–1354. 23 Benkendorf JL, Reutenauer JE, Hughes CA, et al: Patients’ attitudes about autonomy and confidentiality in genetic testing for breastovarian cancer susceptibility. Am J Med Genet 1997;73:296–303. 24 Lerman C, Hughes C, Benkendorf JL, Biesecker B, Kerner J, Willison J, Eads N, Hadley D, Lynch J: Racial differences in testing motivation and psychological distress following pretest education for BRCA1 gene testing. Cancer Epidemiol Biomarkers Prev 1999; 8: 361–367.

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25 Matthews AK, Cummings S, Thompson S, Wolh V, List M, Olopade O: Genetic testing of African Americans for susceptibility to inherited cancers: use of focus groups to determine factors contributing to participation. J Psychosoc Oncol 2000;18:1–19. 26 Hughes C, Fasaye GA, LaSalle VH, Finch C: Sociocultural influences on participation in genetic risk assessment and testing among African American women. Patient Educ Couns 2003;51:107–114. 27 Kinney AY, Croyle RT, Dudley WN, Bailey CA, Pelias MK, Neuhausen SL: Knowledge, attitudes and interest in breast-ovarian cancer gene testing: a survey of a large AfricanAmerican kindred with a BRCA1 mutation. Prev Med 2001;33:543–551. 28 Fouad MN, Partridge E, Green BL, Kohler C, Wynn T, Nagy S, Churchill S: Minority recruitment in clinical trials: a conference at Tuskegee, researchers and the community. Ann Epidemiol 2000;10:S35–S40.

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29 Swanson GM, Ward AJ: Recruiting minorities into clinical trials: toward a participantfriendly system. J Natl Can Inst 1995; 87: 1747–1759. 30 Brown BA, Long HL, Gould H, Weitz T, Milliken N: A conceptual model for the recruitment of diverse women into research studies. J Womens Health Gend Based Med 2002; 9: 625–632. 31 Moorman PG, Newman B, Millikan RC, Tse CK, Sandler DP: Participation rates in a casecontrol study: the impact of age, race and race of interviewer. Ann Epidemiol 1999; 9: 188–195. 32 Abrums M: ‘Jesus will fix it after awhile’: meanings and health. Soc Sci Med 2000; 50: 89–105. 33 Gamble VN: Under the shadow of Tuskegee: African Americans and health care. Am J Public Health 1997;87:1773–1778.

Patterson /Davis /Shelby /McCoy / Robinson /Rao /Banerji /Tomlinson

Community Genet 2008;11:215–223 DOI: 10.1159/000116882

Testing Three Different Cancer Genetics Registry Recruitment Methods with Hispanic Cancer Patients and Their Family Members Previously Registered in Local Cancer Registries in Texas Amelie G. Ramirez a Alexander R. Miller b Kipling Gallion a Sandra San Miguel de Majors a Patricia Chalela a Sandra García Arámburo a a

Department of Epidemiology and Biostatistics, Institute for Health Promotion Research, The University of Texas Health Science Center at San Antonio, and b Cancer Prevention and Risk Assessment Clinic, Cancer Therapy and Research Center, San Antonio, Tex., USA

Key Words Cancer Genetics Network ⴢ Registry ⴢ Recruitment ⴢ Hispanics ⴢ Cancer

tion can have a powerful effect on recruitment. However, more research is needed to determine the cost-efficacy of more labor-intensive approaches to registry accrual. Copyright © 2008 S. Karger AG, Basel

Abstract Objective: To increase accrual among Hispanics to the Cancer Genetics Network national cancer genetics registry. Methods: Drawing from South Texas cancer registries, 444 Hispanic men and women were randomly assigned to one of three experimental conditions: standard direct-mailed procedures (X1), X1 plus culturally tailored materials (X2), and X2 plus interpersonal phone contact (X3). Participants were also surveyed about the effectiveness of the education materials and the phone contact. A refusal survey was provided for those who declined to join the study. Results: A total of 154 individuals joined the Cancer Genetics Network. The X3 condition yielded the greatest accrual (43.2%) compared to X1 (30.9%) and X2 (29.9%; p ! 0.05). Tailored materials appeared to have no effect but were highly regarded. The main reasons for not participating were a lack of interest and time requirements. Conclusion: Interpersonal communica-

© 2008 S. Karger AG, Basel 1422–2795/08/0114–0215$24.50/0 Fax +41 61 306 12 34 E-Mail [email protected] www.karger.com

Accessible online at: www.karger.com/cmg

Introduction

Cancer registries serve an essential role in making cancer data available to researchers who seek to contribute to medicine. Only recently has the literature begun to reflect studies that evaluate the barriers and enabling actions that lead to increased accrual into cancer registries and clinical studies, especially among minorities that are traditionally underreported in medical research. Compounding these shortcomings is a paucity of research on effective methods for improving accrual to registries or trials. In the case of underrepresented populations, for example, ethnic minorities represent 25% of the US population [1], yet they comprise less than 10% of cancer clinical trial participants across the nation [2].

Dr. Amelie G. Ramirez Institute for Health Promotion Research The University of Texas Health Science Center at San Antonio 8207 Callaghan Rd., Suite 353, San Antonio, TX 78230 (USA) Tel. +1 210 562 6500, Fax +1 210 348 0554, E-Mail [email protected]

There is limited cancer registry recruitment among Hispanics, and, not surprisingly, this extends to the recruitment of Hispanics into many clinical trials. This study was conducted to better understand the barriers to Hispanic participation in cancer registries and, to some degree, clinical trial participation. Limited data found in existing literature suggest that key barriers to minority recruitment into clinical trials include lack of education and mistrust of research [3–11], researchers, and the medical system [4, 7–16]. Among others are lack of health insurance [12, 17–19], low income and indirect costs to participation [15–20], and age [21–24]. A comprehensive literature review by Ford et al. [25] has shown that perceived harm of clinical trial participation and fear are among the most frequently reported barriers that minimize clinical trial participation. Some promoters of clinical trial participation among minorities include provider incentives [5, 8, 9], altruism [3, 9, 13, 14, 26], and culturally relevant education about clinical trials [25]. Other studies have shown that interpersonal communications, such as a personal visit or phone call, can increase clinical trial accrual when combined with informational handouts [27–30]. Hughes et al. [29] conducted a literature review of ethnic and racial minority recruitment methods and found that the most common active recruitment methods are via mail (e.g. invitation letter, mailing of survey), telephone, and personal appeal, such as being approached by research staff in clinical settings. The source of the study reported here can be traced to 1998, when the National Cancer Institute funded an innovative cancer family registry, the national Cancer Genetics Network (CGN), providing participating researchers access to a breadth of research data not currently available to most individual cancer genetics programs. The CGN is comprised of eight primary centers around the nation, which are linked to a central informatics center at Massachusetts General Hospital. The Network supports collaborations to investigate the genetic basis of cancer susceptibility, explore mechanisms to integrate this new knowledge into medical practice, and identify ways to address associated psychosocial, ethical, legal, and public health issues [31]. Due to low minority participation rates compared to Caucasian populations in the registry [32], CGN investigators initiated a program to (1) increase minority enrollment in the CGN and (2) contribute to scientific knowledge about enhancement of minority participation in genetics studies. The randomly controlled study described here represents that CGN initiative. This theory-based study examined different com216

Community Genet 2008;11:215–223

binations of educational materials and interpersonal communication to increase rates of accrual to the CGN national registry. Our results indicate that strategic combinations of varying recruitment methods can increase participation in cancer registries.

Subjects and Methods Participants The study recruited Hispanic individuals with a personal and/ or family history of cancer. Subjects were identified from the following four local registries: (1) Cancer Prevention and Risk Assessment Clinic (CPRAC), (2) Familias en Acción Contra el Cáncer (Families in Action Against Cancer – FAMILIAS), (3) Early Detection in Prostate Cancer: San Antonio Center of Biomarkers of Risk for Prostate Cancer (SABOR), and (4) Methodist Hospital Cancer Registry. These four registries either have clinical registrants, research subject registrants, or registrants from hospitals. Participants included English- and Spanish-speaking male and female Hispanics over 18 years of age from San Antonio, Texas, and the surrounding areas. While these registries primarily include patients, the FAMILIAS registry also included 83 family members. Upon merging the registry lists, a few patient overlaps (same patient in two or more registries) were noted; this was corrected by deleting the duplicates. Patients from two registries, CPRAC and the Methodist Hospital, were originally registered by presenting themselves to the clinic/hospital seeking medical services or were referred by a specialist and agreed to have their data included in the corresponding registries. Patients and family members from SABOR and FAMILIAS were recruited from the community to participate in the studies. The actual intervention lasted 5 months. However, preparatory steps, particularly Institutional Review Board (IRB) approvals, were extremely complex and lengthy due to multi-institutional collaborations among health science centers and local hospital registries. In general, the registries made the first contact with a letter notifying registrants of this study and their choice to give the registries permission to share their contact information. Table 1 presents a breakdown of the source, volume, and contact procedures. Although these four registries include more than 26,000 individuals, only about 4,100 qualified as Hispanics through self-identification on the original registry application or determination by surnames. Ultimately, 444 participants responded to the mail-out and accepted the invitation to receive study materials. Experimental Conditions All respondents were randomized, using random digit tables, to one of three experimental conditions (X1, X2, and X3). Table 2 lists the types of materials distributed by condition. All conditions received standard CGN recruitment materials (i.e. CGN pedigree survey and consent documents) by mail. Participants in X2 also received a culturally tailored magazine, while participants in X3 received the magazine and a follow-up telephone call. The magazine, entitled Buena Vida: A Guide to Family Cancer Research, was written in Spanish and English and designed to be culturally and literacy appropriate for a low-literate audience.

Ramirez et al.

Table 1. Study recruitment sources

Source

Total

Hispanic male/female

How identified

Cancer risk

Contact method

FAMILIASa SABORb Methodist Hospitalc

200 1,300 24,000

25/175 285 530

breast prostate all cancers

investigator signs and sends MD signs and sends hospital signs and sends

CPRACd

600

10/120

self-ID self-ID 530 self-ID 3,000 last name self-ID

all cancers

MD signs and sends

a A research study that examined the psychosocial implications of breast cancer genetics among breast cancer survivors and their families. b A prostate cancer early detection and screening research study. c The largest cancer registry data base in the San Antonio area and managed by the Methodist Hospital. d Risk clinic located at the Cancer Therapy and Research Center.

Table 2. Recruitment material distribution

Study materials Study cover letter Consent form (2 copies) Buena Vida magazine Buena Vida magazine evaluation questionnaire Texas Cancer Genetics Consortium questionnaire Refusal questionnaire Self-addressed, postage-paid return envelope Intervention Telephone follow-up

Table 3. CGN registry completion by study condition (n = 444)

X1

X2 X3

! !

! ! ! ! ! ! !

! ! !

! ! ! ! ! ! ! !

The appropriate materials were mailed all together according to each study condition. All materials were available in English and Spanish, and participants received the materials in their preferred language as indicated by them during the recruitment process.

Form and content of the magazine were based on social cognitive theory [33], in which vicarious learning is achieved through the careful application of human social models, whose statements and behaviors illustrate how they have overcome barriers and learned new skills associated with positive health change. Social cognitive theory describes human behavior as an interaction among personal, behavioral, and environmental factors, and humans tend to model behavior that is performed by those with whom they identify. Therefore, the magazine-quoted South Texas Hispanic role models, who speak in local dialect, are presented in familiar environments and engage in typical relationships, discourse, and actions common to those who would receive this material [34, 35]. With photos and quotes of role models and facts regarding cancer and cancer clinical trials, the magazine aimed to raise the audience’s awareness of the risk of cancer and provide information about participation in cancer studies.

Registry Recruitment Methods with Hispanics

Total randomized per condition Complete pedigrees and consent* Response rate, % No/incomplete consent

X1

X2

X3

149 46 30.9 0

147 44 29.9 2

148 64 43.2 6

* p < 0.05.

Phone contact occurred only in the X3 condition and was conducted by trained, bilingual, National Cancer Institute-supported Cancer Information Service personnel located at a regional office in Houston, Texas. Phone operators used cellular phones because calls were made from 8 a.m. to 9 p.m. 7 days a week, and it was evident that the best contact time was between 7:30 and 9 p.m. Phone scripts and response scenarios were developed and practiced prior to performing the follow-up call. Cancer Information Service phone operators also kept logistical records on messages left, call-backs, and other alternative phone numbers to aid in contacting subjects, typically using a cell phone number rather than a home land line. Conditions X1 and X2 were sent materials at the same time. Participants who did not respond within 10 working days of the initial mail-out by returning the study materials were contacted again with a second mail-out. Participants who did not respond within 10 working days of the second mail-out were contacted a third and final time. Participants who returned completed recruitment materials were considered as recruited. A small portion of those who returned completed pedigree surveys failed to include properly completed consent forms, and these were excluded from the study. Table 3 shows the number of individuals per condition who returned study materials. For the X3 condition, the phone operator provided information about the project and offered assistance in completing the pedigree survey and consent documents. Since this condition

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217

Table 4. Demographics of respondents by study condition

Demographic characteristic

Mean age, years Gender Female Male Race White Black/African American Native American/Aleutian/ Eskimo Other Ethnicity Mexican American Puerto Rican Cuban South American Other Spanish Hispanic/Latino NOS Religion affiliation None Catholic Protestant/other Christian Educational level 8 years or less Some high school High school/GED Some college/technical school College graduate or beyond Marital status Never married Married/living as married Separated Divorced Widowed Personal history of cancer Yes No Family history of cancer Yes No

Total

Research condition

(n = 154)

X1 X2 X3 (n = 46) (n = 44) (n = 64)

60.6

61.2

60.6

60.2

40.3 59.7

43.5 56.5

34.1 65.9

42.2 57.8

77.1 0.7

72.7 2.3

82.9 0.0

76.4 0.0

0.7 21.4

0.0 25.0

0.0 17.1

1.8 21.8

53.8 3.4 0.7 1.4 1.4 39.3

54.5 2.3 0.0 0.0 0.0 43.2

51.2 2.4 0.0 1.7 2.4 41.5

55.0 5.0 1.7 1.9 1.7 35.0

2.4 78.0 19.7

2.4 73.2 24.4

0.0 80.6 19.4

4.0 80.0 16.0

2.1 3.4 13.8 35.2 45.5

4.4 4.4 13.3 35.6 42.2

2.4 2.4 14.3 33.3 47.6

0.0 3.4 13.8 36.2 46.6

4.9 71.3 1.4 17.5 4.9

2.3 81.8 2.3 6.8 6.8

2.4 70.7 0.0 19.5 7.3

8.6 63.8 1.7 24.1 1.7

50.3 49.7

54.3 45.7

40.5 59.5

54.2 45.8

66.7 33.3

71.7 28.3

64.3 35.7

64.4 35.6

was more labor-intensive than the other two conditions, materials were sent in batches of 50 every 10 days to enable phone contact within a reasonable time after subjects received the materials. Calls were alternately placed mornings, afternoons, and evenings between 8:00 a.m. and 9:00 p.m., including weekends when necessary. Subjects were called every 2–3 days for a total of 15 times, after which they were categorized as ‘refused to participate’.

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Study Evaluation The main evaluation was the number of people that completed CGN accrual documents. Process data were collected through a call operator-controlled database created to monitor the phone contacts, specifically the frequency of calls, messages left for the subject and call-back information. To ascertain the effects of the culturally tailored magazine and phone contact on subjects, a brief survey was included in the mail-out materials to all subjects. Finally, all conditions also received an even shorter study refusal survey. Only X2 and X3 study participants were asked to complete the magazine survey, since only subjects in these conditions received the publication. The magazine survey that the X2 group received included nine items, while the X3 condition received the same survey with an additional three items that assessed the perceived effectiveness of the phone contact as a recruitment strategy. The short refusal survey explored the reasons for not wanting to complete the CGN materials. Subjects in conditions X1 and X2 were provided one extra question to determine if they would have found telephone contact by study personnel helpful. The analysis included 154 participants who completed all forms. All statistical analysis was performed in 2005 using SPSS for Windows 13.0. Descriptive statistical analysis included frequencies and cross-tabulations. ␹2 analyses were used to assess differences among the three study conditions [36].

Results

Completed CGN pedigree surveys and consent documents were returned by 154 participants, yielding an overall 34.7% response rate (154/444). The underpinning of the operating hypothesis in this study was that culturally tailored materials (contained in X2 and X3 recruitment methods) and the interpersonal follow-up phone call (X3) would lead to greater cancer genetics registry accrual than standard direct-mailed procedures. Due to the exploratory nature of the study, the hypothesis was limited to a general assumption that X3 1 X2 1 X1. Instead, results suggested that X3 1 X1 1 X2 (p ! 0.05). There were 46 registrants from the X1 group (a 30.9% response rate), 44 registrants from the X2 group (a 29.9% response rate), and 64 registrants from the X3 group (a 43.2% response rate). Two respondents in the X1 group and 6 in the X3 group completed the pedigree survey but failed to complete the consent forms, rendering them ineligible for the study. Including those would have boosted the response rate for X3 to 47.3%. Table 4 shows that males outnumbered females in the study, most likely because one of the recruitment sources was a prostate cancer study that yielded 49.0% of the entire study population. Seventy-seven percent of the eligible 154 study respondents reported that they were white, while 21.4% answered race as other. However, Ramirez et al.

100% of the sample acknowledged being Latino, predominantly of Mexican American ethnicity (53.8%). Most subjects were of Catholic faith (78%), and the majority had a high school education or greater, with nearly half having earned a college degree. About 50% of the total sample reported having a personal history of cancer, and 66.7% had a family history of cancer. Considering cancer history across the treatment conditions, subjects in X1 also had slightly higher rates of personal and family cancer history than X2 or X3; however, this did not positively correlate with increased interest in participating in the national CGN cancer registry. Only 5% of all study subjects chose to complete the pedigree and consent documents in Spanish, with the majority selecting English-language materials. Effectiveness of the Magazine One hundred and eight individuals (X2 = 44, X3 = 64) responded to the magazine survey. Table 5 presents response percentages for selected questions. Ninety-nine percent of all survey respondents looked at the magazine, with 68.7% taking between 10 and 15 minutes to review it. All reviewers considered the magazine to be ‘very easy’ to ‘easy’ to understand. Similarly, the majority felt that the examples provided by the social models were ‘somewhat’ to ‘very’ helpful. More than 50% of respondents in both conditions showed the magazine to family members, reflecting further evidence of engagement. The majority in both conditions acknowledged that exposure to the magazine influenced their decision to join the CGN, with 32.6% in X2 and 23.7% in X3 indicating it contributed ‘a lot’. Effectiveness of Telephone Follow-Up All X3 participants were asked questions about the effectiveness of the telephone contact strategy. When asked if having a researcher call and discuss the study helped them make a decision about joining the GCN, 43.1% said it was helpful and 43.1% said it was not. When asked if they would have participated in the study and submitted the forms if they had not been contacted, 58.6% responded affirmatively, although a small percentage (13.8%) said they had not talked with a researcher. Although all subjects in this condition were repeatedly telephoned by call operators, some contacts were never completed after multiple attempts. When X3 subjects were asked whether they would have participated in the study if they had not been called, 27.6% said they would not have joined the study without the call.

Registry Recruitment Methods with Hispanics

Table 5. Magazine/phone survey results

Survey questions

Did you look at magazine? Yes No

Total

Research condition

(n = 108)

X2 (n = 44) X3 (n = 64)

99.0 1.0

100.0 0.0

98.3 1.7

How long did it take you to look at the magazine? 10 min or less 42.2 57.1 15 min 26.5 16.7 20 min 17.6 16.7 30 min 4.9 2.4 More than 30 min 8.8 7.1

31.7 33.3 18.3 6.7 10.0

Did you think the information in the magazine was: Very easy to understand 64.0 70.7 Easy to understand 36.0 29.3 Hard to understand 0.0 0.0 Very hard to understand 0.0 0.0

59.3 40.7 0.0 0.0

In the magazine, did it help you to see what other people, whose lives were affected by cancer, had to say? Very helpful 49.0 53.7 45.8 Somewhat helpful 48.0 41.5 52.5 Not helpful at all 3.0 4.9 1.7 Did you show the magazine to any members of your family? Yes 54.0 52.4 55.2 No 46.0 47.6 44.8 Did the magazine help you to decide about joining the CGN? Yes, a lot 27.5 32.6 23.7 Yes, a little 41.2 44.2 39.0 No, it did not help 31.4 21.4 37.3 Overall, what did you think of the magazine as a way to get information about taking part in cancer studies? Excellent 28.3 32.6 25.0 Very good 41.4 39.5 42.9 Good 29.3 27.9 30.4 Bad 1.0 0.0 1.8 Did having a researcher call and talk to you about this study help you make a decision about joining the CGN registry? n/a 43.1a Yes 43.1a No 43.1a n/a 43.1a Didn’t get call 13.8a n/a 13.8a Did having a researcher from this study answer your questions make you feel better informed about joining the CGN registry? Yes 32.8a n/a 32.8a No 53.4a n/a 53.4a a Didn’t get call 13.8 n/a 13.8a Would you have participated in this study and completed all of the forms if a researcher had not contacted you by telephone? Yes 58.6a n/a 58.6a a No 27.6 n/a 27.6a a Didn’t get call 13.8 n/a 13.8a a

These questions only relate to research condition X3.

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Table 6. Refusal survey results

Survey questions

Discussion Total

Research condition

X1 X2 X3 (n = 59) (n = 22) (n = 25) (n = 12)

Main reason for not participating (check all that apply) Not interested 44.9 44.0 44.8 Don’t read English 1.5 0.0 3.5 Don’t have time 17.4 12.0 10.3 Didn’t understand the forms 1.5 0.0 3.5 Other 34.8 44.0 37.9

46.7 0.0 40.0 0.0 13.3

Did you find the amount of information overwhelming? A lot 33.9 22.7 52.2 18.2 A little 26.8 27.3 26.1 27.3 No 37.5 45.5 21.7 54.5 Not sure/don’t know 1.8 4.5 0.0 0.0 Did you find that there was not enough information in the packet you received? A lot 3.7 9.5 0.0 0.0 A little 11.1 4.8 13.6 18.2 No 79.6 81.0 77.3 81.8 Not sure/don’t know 5.6 4.8 9.1 0.0 Would consider participating in another cancer study in the future? Yes 14.3 9.1 18.2 16.7 No 46.4 59.1 31.8 50.0 Not sure/don’t know 39.3 31.8 50.0 33.3 Would you have been more likely to participate if someone had called you to explain the study? Yes 13.3 13.6 13.0 n/a No 80.0 77.3 82.6 n/a Not sure/don’t know 6.7 9.1 4.3 n/a

Refusal Survey Fifty-nine subjects who chose not to join the CGN took the time to complete and return the refusal survey. The survey, described in table 6, was intentionally brief to reduce response barriers as much as possible. Across all conditions, the primary reason given for not participating was lack of interest or low importance (44.9%). The second greatest barrier was time (17.4%), with the X3 condition showing the greatest support for this category at 40% compared to 12% in X1 and 10.3% in X2. Consistent with the low number of Spanish-language CGN forms completed, language was not cited as a barrier; nor was the amount of information insufficient to make a decision to participate, according to 79.6% of respondents. Finally, 80% of all respondents (77.3% in X1 and 82.6% in X2) said that phone contact would not influence their decision to participate in the CGN. 220

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While the main goal of the project was to determine the efficacy of different recruitment methods to the CGN registry, several other lessons learned bear mentioning here. Future research should carefully factor in the time and other resources necessary to recruit from different registries. Table 1 is included to illustrate this point. Increasing minority recruitment generally requires focusing on racial and ethnic identifiers in clinical records. Therefore, registries that do not request such information or that rely only on last names to identify ethnicity can be problematic. For those researchers who work outside institutions that host registries, it is incumbent upon registry staff to assume significant responsibility for information processing and quality control. In reality, adequate infrastructure may not exist, staff may not be appropriately trained for the task or are overburdened with other tasks, and hiring new staff to support the study may not be feasible. In general, using hospital registries and research studies to identify subjects for recruitment into clinical research can be extremely labor-intensive due to local Institutional Review Boards (IRBs) and Health Insurance Portability and Accountability Act requirements. To make this type of information available would unavoidably require manpower and other resources from host institutions (those that maintain such registries). For example, in this study only hospital personnel could search their records, mail the letters that needed physician signatures, and manage the reception of those letters prior to sending them to the researchers. This extensive data manipulation also required significant quality control, with yet another level of institutional involvement that was beyond the reach of researchers in this study. Finally, regarding preimplementation activities, due to the multi-institutional collaborations that are increasingly necessary in this work, researchers in this study had to work with three different IRBs, each with different procedures of submission, reporting formats, and review. Coordinating this process added 6 months to the project. Though difficult to quantify, recruitment efforts should budget ample time and other resources to properly clear IRB procedures, access registries and ensure sufficient quality control. Our working hypothesis was that culturally tailored materials and interpersonal communication would produce higher accrual rates. This was true for the X3 condition, but, as we found in X2, without call support the tailored materials had little impact on accrual. This was despite the fact that the magazine was considered to be well Ramirez et al.

designed, informative and easy to understand, and 27.5% of subjects that responded to the magazine survey said it was instrumental in their decision to join the CGN. We suspect that those who did not join had more significant barriers than lack of awareness, and the tailored materials were insufficient to overcome those challenges. Barriers may have included response burden caused by the detail required in the pedigree survey and/or apprehension with the legal language and multiple signatures required within the consent documents. According to demographic data, 50% of the sample had a personal history of cancer, yet it is not clear how powerful this was as a motivator for participating in the CGN registry. Nor was language a predictor of lack of participation. While it is possible that low writing/reading skills may have been a barrier for some, this was not the case for those who responded to the refusal survey. Although those who returned the refusal survey represent only a fraction of individuals who chose not to participate, the obvious message from the survey responses is that interest is low. Overcoming this will require more than education about the benefits of clinical trials. Time was also a factor, especially among those who had phone contact. Perhaps general resistance to participation plus the additional time-consuming phone communications with researchers heighten this issue among this group. Regarding the documents themselves, it certainly takes time and effort to read and comprehend consent documents and plot out the different familial relations and their histories as required by the pedigree survey. For some, it meant doing extra work, such as consulting with relatives to answer the questions. Finally, the research activity itself mandates additional consent documents (every survey required a separate consent signature and explanatory paragraph), completing the surveys and mailing them. All of these time-consuming tasks are potential points of resistance that may have further obscured the efficacy of the CGN recruitment strategies. Strengths and Limitations A major strength of this study is that it provides much needed data about different recruitment methods among an underserved population in South Texas. Considering all the institutional requirements, the collaboration among various institutions to bring forth such a large Hispanic contact list to increase the accrual to a national cancer genetics registry could be considered a research milestone. In addition, this paper adheres to the Consolidated Statement of Reporting Trials requirements on randomized controlled trials [37]. Registry Recruitment Methods with Hispanics

With regard to limitations, the study was small, drew on convenience samples residing in local cancer and research study registries, and was intentionally simple in its design. The heterogeneous nature of the original four registries – a clinic, a hospital, and two research studies – contributed to these limitations. It is possible that patients from the hospital/clinic were not as likely to participate in the current study, especially if they had never participated in a research project. Additionally, the contact information from these two registries was less likely to be updated than the contact information from the two more recent research studies. Furthermore, the study was focused on a predominantly Mexican American audience, and it may not be possible to extrapolate similar perspectives and actions on other Latino populations. The consent procedures were onerous due to the multiple surveys and signature requirements, which may have confused or, even worse, repelled some potential study participants. The phone component certainly had the highest response rate; however, it is not clear if the efficacy of this approach is costeffective. The utility of the tailored materials versus phone contact should be further explored, and future research should examine the telephone follow-up intervention by itself to determine effectiveness. Future studies also should not only assess the effects on accrual of a particular recruitment strategy but closely examine the cost issues as well. With the very modest budget allocated for this project, disaggregating costs associated with the overall research activity, quality control measures, human subjects issues, etc., would have been a daunting task.

Conclusion

The results suggest that interpersonal contact via phone can have a beneficial effect on recruitment to cancer genetics registries. The use of culturally tailored materials has been shown to be effective in increasing awareness of health issues, particularly among some audiences. In this study, such materials alone had no appreciable effect on accrual. Clearly, more research is necessary on a plethora of issues covered in this study, from study design through implementation and evaluation. Hopefully, this small experiment can serve as a springboard for more comprehensive inquiry that will lead to improved study accrual practices.

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Acknowledgements The present study was funded by a grant from the National Cancer Institute through the Texas Cancer Genetics Consortium project. It also received partial support from the San Antonio Cancer Institute through grant number 5P30CA54174 and support from faculty and staff of the Redes En Acción Special Populations Network funded by NCI, grant number U01 CA86117. The authors wish to express their gratitude to all four San Antonio,

Texas cancer registries: the Cancer Prevention and Risk Assessment Clinic, Familias en Acción Contra el Cáncer (Families in Action Against Cancer), Early Detection in Prostate Cancer: San Antonio Center of Biomarkers of Risk for Prostate Cancer, and Methodist Hospital. And we thank all of the study participants for their cooperation. The authors also wish to express their appreciation to Dani Presswood and Cliff Despres for assistance in editing the manuscript.

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Community Genet 2008;11:224–233 DOI: 10.1159/000116883

Factors Associated with African Americans’ Enrollment in a National Cancer Genetics Registry C.S. Skinner a J.M. Schildkraut a B. Calingaert a C. Hoyo a S.S. Crankshaw a L. Fish a L. Susswein c C. Jasper d L. Reid b a

Duke Comprehensive Cancer Center, and b North Carolina Central University, Durham, N.C., c Lineberger Cancer Center, Chapel Hill, N.C., d Lombardi Comprehensive Cancer Center, Washington, D.C., USA

Key Words African Americans ⴢ Cancer Genetics Network ⴢ Cancer genetics registry

Abstract This study explored whether reactions to the Cancer Genetics Network (CGN) or CGN enrollment differed by receipt of a standard informational brochure versus a targeted version addressing factors previously associated with African Americans’ health behavior decisions and research participation. The 262 participants, identified through tumor registries or clinic contacts, were mailed brochures and completed phone interviews. When asked whether – based on the brochure – they were or were not ‘leaning toward’ CGN enrollment, about 75% of both standard and targeted groups reported leaning toward. When given the opportunity at the end of the interview, 68% enrolled in the CGN. Trust was strongly related to enrollment. Less education, less satisfaction with cancer care, and individualistic rather than collective orientation were associated with lower trust. Education was also bivariately associated with enrollment, but mediation analysis indicated that the operational mechanism of education’s influence on enrollment was through trust. Copyright © 2008 S. Karger AG, Basel

© 2008 S. Karger AG, Basel 1422–2795/08/0114–0224$24.50/0 Fax +41 61 306 12 34 E-Mail [email protected] www.karger.com

Accessible online at: www.karger.com/cmg

Introduction

Cancer genetics studies must enroll large enough numbers and diverse enough groups of individuals with elevated risks to lead to scientific findings that can be translated into clinical prevention and treatment protocols, but facilitating enrollment of multiple ethnic and racial groups can be a challenge [1–3]. This was certainly the case for the Cancer Genetics Network (CGN) – an innovative cancer family registry funded by the National Cancer Institute in 1998 to provide participating researchers access to a breadth of research data not currently available to most individual cancer genetics programs (e.g. extensive family and personal history data for thousands of individuals). The CGN is comprised of eight main centers around the nation which are linked to a central informatics center. CGN enrollees at these centers complete family and personal history reports and agree to be contacted when there are cancer genetics studies for which they may be eligible. The CGN supports collaborations to investigate the genetic basis of cancer susceptibility, explore mechanisms to integrate the new knowledge into medical practice, and identify ways to address associated psychosocial, ethical, legal and public health issues [4]. C.S. Skinner, MD 2424 Erwin Rd, Ste 602 DUMC 2949 Durham, NC 27710 (USA) Tel. +1 919 684 4791, Fax +1 919 681 4785, E-Mail [email protected]

In its first 3 years, a very small proportion of CGN enrollees represented minority populations. As explained by Bowen et al. [4], 90% were of Non-Hispanic White/ Caucasian ethnicity; remaining participants were 4% Hispanic, 3% African American, 1% Asian, and 2% other ethnicities. In the case of African Americans, at least two factors common to many research studies [2] were contributing to lower enrollment: fewer were invited and, among those invited, a smaller proportion of African Americans than Caucasians enrolled in the CGN. It is still unclear why fewer African Americans are invited to participate in studies and registries similar to the CGN. Factors for nonparticipation among those invited have been a topic of active debate for some time. Few investigations have looked specifically at African Americans’ attitudes regarding cancer genetics registries, but there is some evidence that even African Americans who have previously participated in cancer research have been less likely than Caucasians to enroll in the CGN [5]. In 2003, a group of CGN investigators initiated a program to (1) increase minority enrollment in the CGN and (2) contribute to the scientific knowledge about enhancement of minority participation in genetics studies. This paper presents data from one of the projects in that effort – a trial conducted at Duke University Medical Center, The University of North Carolina (UNC), and Georgetown University Medical Center. With the goal of increasing numbers of African Americans enrolled in the CGN, we developed an informational brochure specifically targeted to African Americans and, via a randomized controlled trial, compared it to the standard brochure used from the CGN’s inception. This paper reports findings about enrollment from the randomized trial and answers to these research questions: (a) Among African Americans who received invitation materials for the CGN: What were reactions to the CGN – overall and by brochure group? and (b) What factors were associated with enrolling/not enrolling in the CGN? Conceptual Model The conceptual model guiding the study, which appears in figure 1, draws from previous research findings that trust is a key determinant of African Americans’ participation in medical research [6]. The figure shows variables expected to be related to trust and, in turn, likelihood to enroll in the CGN. We posited the following. Because trust had been found to be consistently associated with African Americans’ participation in other types of research [7, 8], trust would factor prominently in whether people would opt African Americans’ Enrollment in a National Cancer Genetics Registry

*Demographics Income Education

*Religiosity *Collectivism (family orientation) Experience with health care system

Trust Global Confidentiality Exploitation

Participation

Personal and family history

Fig. 1. Conceptual model. Variables used for brochure targeting are indicated by an asterisk.

to enroll in the CGN. Previous studies had also shown that perceptions of previous medical care influence willingness to participate in research [9, 10]. Therefore, given that most study participants had been treated for cancer at one of the participating institutions, we posited that perceptions of their care would be related to propensity to accept a CGN enrollment invitation from the same institution. We also evaluated whether factors such as collectivism (family orientation) and religiosity – variables that have been shown to be important among African Americans and related to health behaviors [1, 11] – were associated with trust and/or CGN enrollment decisions. Finally, we assessed several demographic variables, and their relationship with trust and with CGN enrollment.

Methods The trial was set up to compare outcomes between participants receiving one of two versions of the CGN informational brochure. We developed a targeted version of the CGN brochure, identified and recruited participants and randomly assigned them to receive the standard or targeted brochure, and followed designated outcomes in both the standard and targeted brochure groups. Brochure Development Factors targeted in the African American version of the CGN informational brochure (fig. 1) include: race, religiosity, collectivism and trust. The targeted brochure was developed and revised based on feedback from a convenience sample of African Americans (employees at Duke University Medical Center and members of a community advisory board from a previous project) who agreed to review the materials and offer suggestions for their improvement [12].

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Brochure Content The resulting targeted brochure was the same size as the standard CGN informational brochure (4-page, 8 1/2 by 11 fold-out) and was printed in the same color scheme as the standard brochure. However, the entire brochure prominently featured African Americans in both photos and text and emphasized it was specifically for African Americans being invited into the CGN. For example: ‘You can help African Americans have a voice in research. Becoming involved in important cancer research can help other African Americans get the best care.’ Section headings were: ‘What you can gain’; ‘What your family can gain’; ‘How you can help’, and ‘Why you should join’. To target family orientation, the brochure was entitled Cancer and Your Family and it included testimonials from African Americans with cancer in their families. A photograph of a woman and her young son accompanied text in which she explained her father died of prostate cancer and she now wonders about the implication of his cancer for her young son: ‘Knowing that cancer runs in your family can help you take care of your loved ones. I don’t think my father ever thought much about cancer in our family. Joining the CGN lets you find out about cancer in your family – and that can help your whole family fight cancer.’ Religiosity and family orientation were both targeted on the back cover via a photograph and testimonial from an African American pastor whose family included several members with colon cancer and who encouraged CGN participation. A portion of the text reads: ‘Having cancer made him think about the cancer in his mother’s family and about losing some of his brothers and sisters to cancer while they were still young … When asked whether he would recommend being in the Cancer Genetics Network, he said “I most certainly would”.’ Trust was targeted by text such as the following that emphasized confidentiality and fidelity of data management: ‘The CGN works hard to keep your information private. They never use your name, or the name of anyone else in the CGN.’ Participant Identification Individuals eligible to participate in this study were identified in three ways: (a) as they checked in for clinic appointments, (b) as they checked in for cancer screening, or (c) by tumor registry. (a) At Georgetown, consecutive patients presenting to be seen in the Medical Oncology Clinic at Lombardi Comprehensive Cancer Center and in oncology clinics at an affiliated hospital during the fall of 2003 were asked to complete a brief CGN screening form outlining their personal and family histories of cancer when they checked in for clinic appointments. (b) The same screening form was also included in the packets of information given to all men who participated in free prostate cancer screenings offered by Lombardi Comprehensive Cancer Center in the fall of 2003. The screening form included an open line on which patients were asked to write in their race and a checkbox to indicate whether someone from the CGN could contact them. Patients whose responses indicated they were Black or African American, who checked the box agreeing to contact, and who met CGN eligibility criteria (personal or family history of cancer) received mailed invitation packets. (c) At Duke and UNC, African American patients with early onset of select cancers – and therefore greater likelihood of having hereditary cancers – were invited to partici-

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pate. Names and contact information were requested from the hospital tumor registries with the following criteria: breast cancer diagnosis ^49 years, colon ^59, ovarian ^60, prostate ^65. The tumor registries include data from all patients treated at the hospitals; they are not subject to bias in voluntary participation. Each institution followed its institutional review board procedures for contacting patients. Both Duke and UNC required physician permission before mailing patients a letter introducing the study’s purpose. Patients could use a toll-free number to request no contact. Whereas Duke physicians were required to sign the letters themselves, letters from UNC were signed by one of the CGN investigators and simply mentioned that the physician had given permission for the contact. Study Procedures The Duke and UNC tumor registries identified 572 individuals who met study eligibility criteria; 423 of these were invited to participate. The 149 who were not invited comprised 123 for whom physicians did not sign and return invitation letters, 4 who could not be contacted, 2 who upon contact were determined to not meet eligibility criteria, and 17 for whom contact was not attempted because enrollment quota was already met. At Georgetown, research assistants (RAs) collected 183 screening forms from eligible individuals. Of these, 122 marked the check box on the form indicating permission to receive study information. As shown in figure 2, the 545 individuals identified as potential participants (423 from Duke/UNC and 122 from Georgetown) were randomly assigned to receive either targeted (n = 272) or standard (n = 273) CGN brochures, along with notice that they would receive a telephone call inviting them to participate in the study and receive a USD 10 gift certificate for doing so. Within each of the three sites – Duke, UNC and Georgetown – assignment to receive the standard or targeted brochure came via a computer-generated random allocation sequence. At least 1 week following letter/brochure mailing, RAs blinded to brochure group assignment attempted to conduct computer-assisted telephone interviews with individuals who had read at least part of the brochure. For those who wanted to participate but had not read any of the brochure, RAs arranged to send another copy (if needed) and scheduled an interview time convenient to the participant. Of those assigned to receive the targeted African American brochure, 122 (45%) consented and completed the telephone interview whereas 150 (55%) declined; of those assigned to receive the standard CGN brochure, 140 (51%) consented and completed the interview; 133 (49%) declined to participate (fig. 2). The interview began by assessing whether, based on what he or she read in the brochure, the participant was or was not ‘leaning toward’ enrolling in the CGN and then assessed attitudes and beliefs, included in the study’s conceptual model (fig. 1) that might influence propensity to enroll. At the end of the interview, individuals were given the opportunity to enroll in the CGN Registry. As explained by Bowen et al. [4], CGN enrollment involved completing an extensive family and personal history report and agreement to be recontacted to update information and/or be invited into future cancer genetics studies for which they were likely to be eligible. Therefore, enrollment in the CGN involved collection of many more data than the study reported here, as well as commitment to longitudinal follow-up. Those who opted for enrollment scheduled an appointment for CGN telephone data collection.

Skinner /Schildkraut /Calingaert /Hoyo / Crankshaw /Fish /Susswein /Jasper /Reid

423 Individuals with early age at Dx of cancer identified through Duke and UNC Tumor Registries and 122 self-referrals from Georgetown Prostate Screening program with personal or family history of cancer

Randomly assign 545 of these to receive standard or targeted brochure and invitation to Attitudes study Targeted brochure = 272 (272/545 = 50%)

Standard brochure = 273 (273/545 = 50%)

Call to invite participation in Attitudes about participating in the CGN study

Yes, verbal consent obtained and survey administered = 262 (262/545 = 48%) Standard = 140 (140/273 = 51%)

No, decline participation = 283 (283/545 = 52%) Standard = 133 (133/273 = 49%)

Targeted = 122 (122/272 = 45%)

Invite to enroll in Cancer Genetics Network Registry

Targeted = 150 (150/272 = 55%)

STOP

178 enrolled in CGN (178/262 = 68% of those who participated in Attitudes study) (178/545 = 33% of total invited) Standard = 95 (95/140 = 68% of those in Attitudes study) (95/267 = 35% of total invited)

Targeted = 83 (83/122 = 68% of those in Attitudes study) (83/272 = 31% of total invited)

Fig. 2. Participant flow chart.

Measures Constructs included in our conceptual model (fig. 1) were measured in the telephone interview and are described below. Reliability data are reported for the scales; validity data were not available prior to the study. Patient characteristics, including age, education, employment, cancer family history, and perceived health were measured via standard items from the CGN’s core data elements. Cultural factors of religiosity and family orientation were assessed via abbreviated versions of scales developed by Lukwago et al. [13] and validated in a large population of African Americans. These abbreviated versions used ‘top-loading’ items from factor analyses, indicating that the selected items were most strongly correlated with each other. All scales used 5-point Likert-type responses, with higher summed scores denoting higher levels. The following items measuring religiosity had a Cronbach’s ␣ of 0.79: (1) ‘You are often aware of the presence of God in your life’; (2) ‘You have a personal relationship with God’; (3) ‘When you are ill, you pray for healing’; and (4) ‘You pray often’. Collectivism (sometimes referred to as ‘family orientation’) was measured via four items which, in our sample, had Cronbach’s ␣ of 0.85. Using a 5-point scale from ‘not at all important’ to ‘very important’, participants answered the questions, ‘How important is it that you and your family: (a) turn to each other in times of trouble?

(b) raise each other’s children whenever there is a need to do so? (c) do everything you can to help each other move ahead in life? and (d) take responsibility for caring for older family members?’ Trust was measured via a new scale developed for this study to address three components of trust identified in the literature [14] – global trust, confidentiality, and exploitation. Items, drafted by the research team, were reviewed by CGN investigators and research staff at participating institutions, tested in a convenience sample of African American workers at UNC and Duke, and evaluated by members of a Health Education class composed of predominately African American students at North Carolina Central University. The resulting four items were: (a) ‘You do not believe that your information would be kept private’; (b) ‘If you answer questions about cancer, someone might see your answers and use this information against you’; (c) ‘You just don’t trust researchers’; and (d) ‘You think being in the Cancer Genetics Network might lead to discrimination from groups like Medicaid or insurance companies’. Five-point Likert-style responses ranged from (1) ‘strongly agree’ to (5) ‘strongly disagree’, where disagreeing with the items indicates more trust. Reliability coefficient (Cronbach’s ␣) for these items was 0.72. We summed responses to create a score with higher values representing higher levels of trust. Perceived barriers and benefits for registry participation (i.e. reasons to/not to enroll) were assessed first in open-end and later

African Americans’ Enrollment in a National Cancer Genetics Registry

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Table 1. Participant demographics, by university

Age, median (range), years Gender Female Male Education Graduate education College graduate Some college Vocational/business High school graduate 2 1st or 2nd degree relatives with cancer 0 1 2 >2 Enrolled in CGN

Duke (n = 97)

Georgetown (n = 57)

UNC (n = 108)

Overall (n = 262)

60 (25–91)

53 (37–77)

52 (32–72)

31 (32) 66 (68)

28 (49) 29 (51)

71 (66) 37 (34)

130 (50) 132 (50)

<0.001

15 (15) 22 (23) 6 (6) 9 (9) 21 (22) 24 (25)

17 (30) 11 (20) 17 (30) 0 (0) 8 (14) 3 (5)

14 (13) 12 (11) 28 (26) 6 (6) 28 (26) 20 (19)

46 (18) 45 (17) 51 (20) 15 (6) 57 (22) 47 (18)

<0.001

2 (2) 5 (5) 14 (14) 25 (26) 49 (51) 2 (2)

36 (64) 4 (7) 9 (16) 0 (0) 1 (2) 7 (13)

0 (0) 3 (3) 56 (52) 0 (0) 34 (31) 14 (13)

38 (15) 12 (5) 79 (30) 25 (10) 84 (32) 23 (9)

<0.001

39 (40) 31 (32) 17 (18) 10 (10)

9 (16) 30 (53) 16 (28) 2 (4)

42 (39) 37 (34) 22 (20) 7 (6)

90 (34) 98 (37) 55 (21) 19 (7)

0.088

15 (15) 29 (30) 19 (20) 34 (35) 70 (72)

5 (9) 10 (18) 13 (23) 29 (51) 38 (67)

22 (20) 25 (23) 20 (19) 41 (38) 70 (65)

42 (16) 64 (24) 52 (20) 104 (40) 178 (68)

0.161

56 (25–91)

p value <0.001

0.518

Figures in parentheses indicate percentages, unless otherwise indicated.

in close-end form. Open-end questions asked ‘What are reasons you would not want to be listed in this registry of individuals who have developed cancer at a young age?’ and ‘What are the reasons you would want to be listed in this registry?’ A list of ‘silent code’ potential responses (e.g. not read aloud, but used by interviewers to code responses) included previous reasons offered by CGN ‘decliners’ and those identified from the published literature. Interviewers used the silent code, when appropriate, and recorded, verbatim, those not listed on the silent code. For each response given, the interviewer asked and recorded the answer to, ‘Is that a very important reason or a somewhat important reason for you?’ Closed-end measures of perceived barriers and benefits, or reasons not to/to participate in the CGN, were each measured via items with Likert-style responses ranging from strongly disagree to strongly agree. However, the items cannot be considered together as a summed scale because Cronbach’s ␣ was only 0.61 for barriers; 0.44 for benefits. Barrier items were: (a) ‘There’s nothing in it for you or your family’; (b) ‘You do not feel comfortable talking to a researcher about cancer in your family’; (c) ‘Being in the Cancer Genetics Network would make you think too much about

228

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cancer’; and (d) ‘Being in the Cancer Genetics Network would take too much time’. Benefits items were: (a) ‘Your being in the CGN could help researchers in the fight against cancer’; (b) ‘You want to make sure the CGN finds information that helps African Americans’; and (c) ‘Being in the CGN will help you learn important information about cancer’. Statistical Methods Statistical analyses were conducted in SAS v8.2 (Statistical Analysis Software, Cary, N.C., USA) using contingency tables and unconditional logistic regression to test whether demographics, religiosity, collectivism, experience with the health care system, or personal and family history of cancer were associated with CGN intention to enroll and actual enrollment. Bivariate analyses were conducted to identify response differences between study groups – ␹2 tests of association for discrete outcome measures and ANOVAs and t tests for nonparametric analogues. When significant associations were detected, we used path analysis to determine whether the associations were mediated by trust. Predictors of participation were identified using logistic regression models.

Skinner /Schildkraut /Calingaert /Hoyo / Crankshaw /Fish /Susswein /Jasper /Reid

Table 2. Perceived barriers to CGN Overall (n = 262)

Leaning towards

Enrolled in CGN

Education

Gender

yes other p value (n = 196) (n = 66)

no yes p value (n = 84) (n = 178)

≤HS (n = 104)

>HS p value (n = 157)

female (n = 130)

Age male p value (n = 132)

≤55 (n = 128)

>55 p value (n = 134)

Q16a: There’s nothing in it for your family 10 8 18 0.020 8

11

0.522

14

20

0.060

5

15

0.014

6

14

0.043

Q16b: Don’t feel comfortable talking 16 11 30 0.001

19

15

0.371

30

7

0.000

13

19

0.239

14

18

0.406

Q16c: Don’t believe info kept private 37 34 48 0.039

48

33

0.021

42

34

0.191

35

40

0.374

39

36

0.611

Q16d: Someone might use info against you 24 22 32 0.135 27

23

0.446

27

22

0.460

25

23

0.774

30

19

0.062

Q16e: Don’t trust researchers 18 15 27 0.027

15

0.057

27

11

0.002

18

18

1.000

19

17

0.750

Q16f: Might lead to discrimination by insurances 26 22 36 0.033 30 24

0.288

29

24

0.385

28

24

0.481

27

24

0.573

Q16g: Might think too much about cancer 21 15 36 0.001 30

16

0.014

35

11

0.000

20

21

0.879

19

22

0.542

7

0.000

18

9

0.036

8

17

0.062

12

13

0.713

Q16h: Take too much time 13 6 32 0.000

25

24

Figures reflect percentage of those who agree or strongly agree with each statement.

Results

Descriptive Results Table 1 presents numbers of participants and their characteristics, by study center. Participants were evenly distributed by gender at Georgetown, whereas females were overrepresented at UNC (66%) and underrepresented at Duke (32%). Participants were significantly older at Duke (median = 60 years) than at Georgetown or UNC (medians 53 and 52 years, respectively). Due to differential recruitment strategies, most (64%) participants at Georgetown reported not having ever had cancer, whereas all the Duke and UNC participants – identified via hospital tumor registry – had been diagnosed with cancer. Most common cancer types at Duke were prostate (51%) and kidney (26%), whereas participants recruited from UNC were most likely to have had breast (52%) or prostate cancer (31%). Two thirds of participants reported having a firstdegree relative with cancer, with the Georgetown sample having the highest percentage (84%). Overall enrollment in the CGN was 68% and was similar across the three study centers. Random assignment was successful in that all demographic characteristics were similar between those who received standard and targeted brochures. African Americans’ Enrollment in a National Cancer Genetics Registry

What Were Reactions to the CGN – Overall and by Brochure Type? We measured inclination toward CGN enrollment: (1) at the beginning of the telephone interview, participants were asked if – based on their impressions from the brochure – they were or were not leaning toward enrolling in the CGN; (2) at the end of the interview, study participants had the opportunity to actually enroll or not enroll in the CGN. Findings at each of these points – overall and by brochure type – are reported below. Reactions by Brochure Type Although we had expected more positive reactions among participants who had received the targeted rather than the standard CGN brochure, reactions from targeted and standard recipients were similar in every way measured. Percentages of standard CGN brochure recipients who said they were ‘leaning toward’ participation, ‘not leaning toward’, and ‘don’t know’ were 72, 14 and 14%, respectively. For targeted brochure recipients, the corresponding percentages were 78, 12 and 10%. We analyzed questionnaire responses to see if there were any differences between standard vs. targeted brochure groups in any of the variables of interest – inclinations toward enrolling in the CGN, trust, perceived reasons for and Community Genet 2008;11:224–233

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Table 3. Questionnaire responses, history of cancer, and demographics, by CGNenrollment

CGN choice

Wilcoxon

enrolled (n = 178)

declined (n = 84)

Nothing in it for my family Don’t feel comfortable Make think too much about Too much time

19.381.8 19.780.6 15.784.0 1.681.2 1.781.3 1.881.3 1.781.0

19.382.0 19.581.9 14.084.4 1.781.1 1.881.4 2.381.6 2.581.3

0.807 0.829 0.003 0.356 0.459 0.046 0.000

Benefits

Help researchers fight cancer Help African Americans Help learn info about cancer

4.880.6 4.880.7 4.780.6

4.680.8 4.880.7 4.680.8

0.011 0.991 0.284

Experience with care1

Overall care rating Doctors did whatever it took Doctors were thorough

4.780.8 4.880.6 4.880.6 54.589.8 1.181.0 2.582.2 153 (86%) 24 (14%) 33 (19%) 35 (20%) 38 (21%) 9 (5%) 35 (20%) 28 (16%) 94 (53%) 84 (47%)

4.581.0 4.880.8 4.780.8 55.6810.5 1.081.0 2.181.8 69 (82%) 15 (18%) 13 (16%) 10 (12%) 13 (16%) 6 (7%) 22 (27%) 19 (23%) 36 (43%) 48 (57%)

0.312 0.995 0.532 0.390 0.602 0.193 0.364

Religiosity scale Collectivism Trust Barriers

Age, years 1st degree relatives with cancer 1st or 2nd degree relatives Personal history of cancer (Q19) Education

Gender

Yes No Graduate education College graduate Some college Vocational/business High school graduate/GED
0.033

0.133

Values are expressed as mean 8 SD, unless otherwise indicated. Limited to those who were treated for cancer (154 enrolled, 70 declined).

1

against enrolling in the CGN, and even reactions to the brochures. There were no differences between groups at the ␣ = 0.05 significance level. Overall Reactions Among all study participants, 75% (n = 196) reported that, just having gotten the brochure with information about the CGN, they were leaning toward being in it (table 2); 13% were not leaning toward (n = 34) and 12% were not sure (n = 31). When asked the open-end question about why they were leaning toward enrollment, responses were: to help self/family/others (n = 151); good way to learn information (n = 55); important for African Americans (n = 13); because of cancer in self-family (n = 13); would be interesting (n = 11); curious about the study (n = 10); other (n = 18). 230

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Open-end responses about why they were not leaning toward enrollment included: would take too much time (n = 8); not want to think about cancer (n = 6); not enough information to make a decision (n = 6); not understand what study is about (n = 5); concern about privacy (n = 5); no advantage/benefit/reason to enroll (n = 3); not kind of person CGN is looking to enroll (n = 2); other (n = 11). Although, as shown above, only 5 participants mentioned concern for privacy in response to the open-end question, such concerns were the most frequently endorsed items in closed-end assessments of perceived barriers to CGN enrollment (table 2). Not believing that one’s CGN information would be kept private and agreeing that CGN participation might lead to discrimination were both significantly associated with not leaning toSkinner /Schildkraut /Calingaert /Hoyo / Crankshaw /Fish /Susswein /Jasper /Reid

ward being in the CGN (p = 0.039 and 0.033, respectively). Another concern mentioned by more than a fifth of participants – that being in the CGN would make them ‘think too much about cancer’ – was also associated with leaning toward or against enrollment (p = 0.046) and was a significantly more likely response among participants with less than a high school education (p ! 0.0001; data not shown). There was overwhelming agreement with each of the three benefit items (94–96%).

p = 0.033 p = 0.102

CGN enrollment

Education

p = 0.011 Trust

p = 0.003 p = 0.010

Fig. 3. Significance of relationships between education, trust and

enrollment. Statistics in bold are multivariate.

What Factors Were Associated with Enrolling/ Not Enrolling in the CGN Registry? We had expected greater CGN enrollment among recipients of the targeted brochure. However, enrollment did not differ by brochure type – exactly 68% of each brochure group went on to join the CGN. Leaning toward enrollment at the beginning of the interview seemed to predict ultimate enrollment; 75% of standard brochure recipients who had been ‘leaning toward’ CGN enrollment and 79% of targeted brochure recipients ‘leaning toward’ enrollment actually did join the CGN. As illustrated in table 3, higher trust, lower perceived barriers, and higher education were all significantly associated with enrollment. With higher scores indicating more trust, mean trust scale score for those who enrolled in the CGN was 15.7 compared to 14.0 for those who did not enroll (p = 0.004). Education level was also significantly associated with enrollment (p = 0.033; table 3). A total of 60% of those who enrolled had at least some college education (21% had some college, 20% were college graduates, and 19% had more than college) compared to a total of 40% of those who declined enrollment (16% had some college, 12% were college graduates, and 16% had more than college education). Responses to two of the four barrier questions differed significantly between those who did/did not enroll; those who declined to enroll were more likely to respond that enrolling in CGN would ‘make them think too much about cancer’ (p = 0.046) or ‘take too much time’ (p ! 0.001). There were no significant associations between participation and religiosity, collectivism, opinion of the health care received, personal or family cancer history, age, or gender. These findings confirmed expectations that trust would be related to CGN enrollment. But, among the other variables we explored – demographics, religiosity, collectivism, experience with the health care system, and cancer history – only education was significantly associated with enrollment. Therefore, we assessed whether the operational mechanism of education on enrollment might be mediated through trust. And, because there African Americans’ Enrollment in a National Cancer Genetics Registry

was indeed a strong association between education and trust (p = 0.011), we tested whether trust mediated the effect of education on enrollment. As shown in figure 3, with both trust and education included in the model, the effect of education was no longer significant (p = 0.102) but trust remained significantly related to enrollment (p = 0.010). To identify factors associated with trust, we performed bivariate analyses (data not shown). Variables significantly and positively associated with trust included: collectivism summary score (p = 0.011), satisfaction with cancer care (p = 0.010), and education (p = 0.011). We then conducted a multivariate analysis with the 216 participants with complete data, using a stepwise regression to determine which variables (age, gender, education, religiosity summary score, collectivism summary score, experience with the health care system, and cancer history) were associated with trust. Findings identified the same variables that were significant in the bivariate analyses; significance of associations was virtually identical too (education was p = 0.009, collectivism was p = 0.012, and satisfaction with cancer care was p = 0.016). Post-Study Observational Cohort At the end of the study, the UNC and Duke sites obtained institutional review board approval to extend the trial by following an observational cohort of African Americans identified via the hospital tumor registries. These individuals (n = 87) were randomly assigned to receive targeted or standard brochures and then invited to enroll in the CGN registry; they were not invited into the study assessing attitudes about the CGN, asked any questions about their opinions, or provided with remuneration. CGN enrollment under this observational condition was similar to that of the study population (63 and 68%, respectively). And, as was the case among study participants, enrollment rates were not higher among targeted brochure recipients. Community Genet 2008;11:224–233

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Discussion

This study, undertaken as a result of previously low enrollment in the CGN among African Americans, sought to explore overall reactions to the CGN and determine whether those reactions, as well as CGN enrollment, differed among those who received the standard brochure that had been used since the CGN’s inception versus a targeted brochure developed specifically for African Americans. The targeted brochure addressed factors that had been shown in previous research to be associated with health behavior decisions and research participation. Potential participants were identified through tumor registries or clinic contacts. Findings in this randomized controlled trial showed no between-group differences in reactions to the brochure or the CGN, or in actual decisions to enroll in the CGN. Readers should note that the study was not conducted among a population-based sample and therefore findings cannot be generalized to the universe of all African Americans who might be invited to participate in a cancer genetics registry. There were three methods for recruitment; the first two generated a convenience sample by inviting all eligible African Americans who presented for clinical appointments or no-cost cancer screenings. We do not have information about how many of these patients were eligible but were not included. The third method identified eligible persons via hospital tumor registries and invited all for whom we obtained signed invitation letters from their physicians. Via this method, there were 123 identified patients for whom we did not obtain signed invitation letters from their physicians; available data on gender and cancer type (not shown) for these show the proportions of women and men for whom we obtained signed invitation letters did not differ significantly. However, there was a difference by two cancer types, in that signed physician invitation letters were obtained for a larger proportion of breast cancer patients and a smaller proportion of kidney cancer patients. It may be that physicians considered their breast cancer patients to be more healthy and able to participate than the patients who had been treated for kidney cancers. A further limitation of the sample is that, even among the 545 individuals identified and to whom the brochure was mailed, we were able to contact only 354 by phone and, of these, more than a quarter (92) declined participation. All subsequent findings must therefore be considered as representing only the 74% who agreed to participate and completed telephone interviews. Although little informa232

Community Genet 2008;11:224–233

tion exists about those who refused to complete the telephone interview, it is likely that those least interested in or most opposed to research declined participation. Study findings clearly showed that brochure type affected neither study participation nor reactions to the CGN. Recipients of the targeted brochure were no more likely to agree to participate in the study than were standard brochure recipients. Of those who participated in the study, most reported that their first impression conveyed by the brochure left them ‘leaning toward’ enrolling in the CGN. Of those reporting ‘leaning toward’, about three quarters of the standard and targeted brochure groups actually did enroll. What were respondents’ perceived reasons for enrolling in the CGN? Most common answers to this open-end question were a desire to help others and to learn more about cancer genetics. A similar open-end question regarding reasons not to participate elicited fewer responses; however, respondents did indicate some barriers to participation in response to close-end items. The most commonly endorsed responses concerned privacy and discrimination, and endorsement of these barriers was significantly inversely related to ‘leaning toward’ enrollment. Having found that type of brochure was not related to either perceptions about the CGN or actual enrollment, we investigated which factors were related to enrollment. Findings validated the initial prediction that trust and perceived barriers would be significantly associated with enrollment. However, because recipients of the brochure developed to target these factors were not significantly different from standard brochure recipients in attitudes toward the CGN or enrollment, the targeted brochure seems to have affected neither trust nor perceived barriers. This is not surprising, given that it is difficult to address potentially deep-seated perceptions with a simple brochure, especially among less educated groups for whom print may not be an effective medium (e.g. lower education was significantly related to lower enrollment). We had tested the targeted brochure because (a) it was an inexpensive strategy that, if effective, would have been widely ‘disseminable’ and (b) studies among similar populations had found that using print media that address factors in our model can be effective in changing attitudes and facilitating health behavior change [3]. However, it may be that trust – a major factor associated with enrollment – is more difficult to influence than other attitudes and therefore less amenable to change via a print message. An important contribution of this study is the 4-item trust scale that had a strong reliability coefficient (Cronbach’s ␣ = 0.72). There is evidence for the trust scale’s Skinner /Schildkraut /Calingaert /Hoyo / Crankshaw /Fish /Susswein /Jasper /Reid

validity in that scores were, as predicted, strongly related to enrollment. Given that trust had such an influence on CGN enrollment, we explored which other factors in the model were related to trust. Findings indicated that less education, less satisfaction with cancer care received at the medical institution through which the study was being conducted, and a more individualistic rather than a collective (or family) orientation were associated with lower trust. Education was also bivariately associated with enrollment, but mediation analysis indicated that the operational mechanism of education’s influence on enrollment was through trust. Because CGN enrollment was higher than we had expected, we wondered whether enrollment had been enhanced by the participants’ experience of being invited to share their attitudes about being in the CGN and by having been remunerated for their study participation. To investigate these questions in a post-hoc manner, investigators at Duke and UNC conducted a small observational study by following an additional cohort of African Americans who were identified via tumor registries and randomly assigned to receive targeted or standard brochures but not invited into the study assessing attitudes about the

CGN or provided with remuneration. The facts that (1) CGN enrollment under this post-hoc observational condition was similar to that of the study population and that (2) enrollment rates were not higher among targeted brochure recipients suggest data collection and remuneration did not meaningfully alter either the brochures’ effect or individuals’ propensity to enroll in the CGN. Study findings confirmed previous reports that lower enrollment rates among African Americans can be at least partly explained by fewer African Americans being identified and invited for participation [2]. Purposefully identifying and inviting African Americans into the CGN registry led to enhanced enrollment rates. Results from the observational cohort suggest that CGN enrollment was not a function of being asked opinions or receiving remuneration for study participation. Findings from both the main study and the post-hoc observational cohort indicate that enrollment was not influenced by whether invitation materials were targeted to address cultural factors shown to be important among African Americans. This study confirmed that trust is a key issue in participation decisions; if this is the case among study participants, it is likely also true for the sizable proportion of potential subjects who did not agree to participate in this study.

References 1 Ard J, Skinner CS, Svetkey L: Acculturation and dietary intake among African American participants in the PREMIER trial. J Behav Med 2005;28:239–247. 2 Hughes C, Peterson SK, Ramirez A, Gallion KJ, McDonald PG, Skinner CS, Bowen D: Minority recruitment in hereditary breast cancer research. Cancer Epidemiol Biomarkers Prev 2004;13:1146–1155. 3 Kreuter M, Skinner C, Holt C, Clark E, Haire-Joshu D, Fu Q, Booker A, Steger-May K, Buckholtz D: Cultural tailoring for mammography and fruit and vegetable intake among low-income African American women in urban public health centers. Prev Med 2005;41:53–62. 4 Bowen DJ, Vu T, Kasten-Sportes C: Increasing minority participant enrollment into a cancer family registry: the Cancer Genetics Network. Community Genet 2008;11:191– 192.

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10 Mouton CP, Harris S, Rovi S, Solorzano P, Johnson MS: Barriers to black women’s participation in cancer clinical trials. J Natl Med Assoc 1997;89:721–727. 11 Lukwago SN, Kreuter MW, Holt CL, StegerMay K, Bucholtz DC, Skinner CS: Sociocultural correlates of breast cancer knowledge and screening in urban African American women. Am J Pub Health 2003;93:1271–1274. 12 Skinner C, Kreuter M, Buchanan A, Holt C, Buckholtz D: Adaptation of tailored intervention message libraries. Health Educ 2003; 103:221–229. 13 Lukwago SN, Kreuter MW, Bucholtz DC, Holt CL, Clark EM: Development and validation of brief scales to measure collectivism, religiosity, racial pride, and time orientation in urban African American women. Fam Community Health 2001;24:63–71. 14 Hall MA, Dugan E, Zheng B, Mishra AK: Trust in physicians and medical institutions: what is it, can it be measured, and does it matter? Milbank Q 2001;79:613–639.

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Community Genet 2008;11:234–240 DOI: 10.1159/000116884

Testing Targeted Approaches to Enhance Cancer Genetics Network Minority Recruitment within Asian Populations Lari Wenzel a Deborah Bowen d Rana Habbal b Nancy Leighton b Thuy Vu c Hoda Anton-Culver b a

Center for Health Policy Research, b Epidemiology Division, School of Medicine, University of California, Irvine, Calif., c Fred Hutchinson Cancer Research Center, Seattle, Wash., and d Social and Behavioral Sciences, School of Public Health, Boston University, Boston, Mass., USA

Key Words Cancer genetics ⴢ Minority recruitment ⴢ Cancer Genetics Network

Abstract Background/Aims: Asian Americans have been underrepresented in cancer research. The purpose of this study was to evaluate the efficacy of a multiple arm recruitment approach in improving Asian recruitment into the Cancer Genetics Network (CGN). Methods: 1,096 potential participants, identified through cancer registries located at University of California, Irvine (UCI) and Fred Hutchinson Cancer Research Center (FHCRC), were randomly assigned to receive one of four recruitment approaches. Results: A 6.2% gain in Asian participation into the CGN was achieved over a 2-year period at FHCRC and UCI, which contributed a 2% CGN-wide increase in overall Asian enrollment. Site-specific differences in recruitment success by study arm were observed. Conclusion: Novel recruitment approaches can assist in improving recruitment of Asian populations into cancer genetic research studies. Copyright © 2008 S. Karger AG, Basel

© 2008 S. Karger AG, Basel 1422–2795/08/0114–0234$24.50/0 Fax +41 61 306 12 34 E-Mail [email protected] www.karger.com

Accessible online at: www.karger.com/cmg

Introduction

Advances in human genetics have provided an important new opportunity to identify cancer genes through studies of cancer-prone families and individuals with no family history of cancer. However, recruitment into cancer research trials, including cancer genetics studies, has traditionally yielded large percentages of non-Hispanic Caucasians, with very little representation by minority populations. Although there is an urgency to continue cancer genetics research, additional observational and experimental studies are especially needed to evaluate the effects of different recruitment strategies on enrollment decisions among ethnic and racial minorities [1]. Barriers to recruitment of minority populations can affect the generalizability and impact of research findings for those populations [2, 3]. Although Asian Americans represent one of the major minority groups in the US, there is a paucity of published literature concerning the participation of Asian Americans in cancer research [3–6]. Many recruitment barriers exist among the Asian population [4–9], generally including a lack of knowledge related to cancer and genetics, as well as cultural beliefs which influence attitudes toward cancer. For example, Lari B. Wenzel, PhD University of California, Irvine Center for Health Policy Research, 111 Academy Way, Suite 220 Irvine, CA 92697 (USA) Tel. +1 949 824 3926, Fax +1 949 824 3388, E-Mail [email protected]

beliefs that cancer is a curse, a punishment from God, and is contagious, promotes behaviors of avoidance and denial rather than proactive health-seeking behavior [10, 11]. Other commonly reported barriers to participation in research among Asians include language problems, lack of culturally relevant cancer information, complex protocols, fear of ill effects, fear of experimentation or belief that experimental protocols are inferior to standard care, and influence of family in decision-making [4–6]. In addition, Vietnamese individuals may be reluctant to disclose personal health information fearing that the information may be used against them, or fearing that they will lose face within their family and community [Ghosheh et al., pers. commun.]. Orange County, California is home to the largest Vietnamese population outside of Vietnam. A local pilot study of Vietnamese-serving health care practitioners in Orange County, California, reinforced concepts that cultural attitudes and beliefs strongly influenced perceptions of cancer and genetics [Ghosheh et al., pers. commun.]. When queried about specific recruitment barriers, the following were prominent: (1) distrust in research; (2) language; (3) desire to stay only within their own community. The following were recommended actions to reduce recruitment barriers: (1) recruit from credible, trusted community leaders; (2) use grass roots community organizations; (3) sponsor local community dinners; (4) recruit through local Vietnamese radio. Anecdotally, it is worth noting that all practitioners stated that our standard population-based mail-out recruitment method would not be effective with Vietnamese subjects. In 1998, the National Cancer Institute funded an innovative cancer family registry: the national Cancer Genetics Network (CGN) which provided participating researchers access to a breadth of research data not currently available to most individual cancer genetics programs. The CGN is comprised of eight main centers around the nation which are linked to a central informatics center. The Network supports collaborations to investigate the genetic basis of cancer susceptibility, explore mechanisms to integrate this new knowledge into medical practice, and identify ways to address associated psychosocial, ethical, legal, and public health issues [12]. Early recruitment efforts into the CGN did not specifically target minorities; thus, minority participation rates lagged behind those of Caucasian families [Bowen, Vu and Kasten-Sportes, this issue, pp. 191–192]. As of May 2002, the CGN contained data on 15,007 participants and 241,948 family members. The majority of CGN participants were of Non-Hispanic White/Caucasian ethnicity Minority Recruitment within Asian Populations

(90%), with few numbers of Hispanic (4%), Black (3%), Asian (1%), and other ethnicities (2%). Therefore, the CGN investigators initiated a program (1) to increase minority enrollment in the CGN and (2) to contribute to the scientific knowledge about enhancement of minority participation in genetics studies. This study presents data from one of the projects in that effort. Since Asians make up more than 7% of all cancers diagnosed in the states of Washington and California, it was essential to work toward a greater representation of this population in a multi-ethnic registry. The study described herein offered an opportunity to examine relative benefits of four mail-out recruitment strategies, thereby altering the customary populationbased recruitment approach in order to potentially increase Asian participation in the CGN. The purpose of this paper was to evaluate the efficacy of a multiple-arm recruitment approach conducted through cancer registries located at University of California, Irvine (UCI), and Fred Hutchinson Cancer Research Center (FHCRC), and evaluate the relative efficacy of each arm in enhancing Asian recruitment to the CGN.

Materials and Methods Conceptual Framework for Recruitment Approaches Since the decision to accept or decline participation in an initial survey to collect epidemiological and medical history information is similar to decisions to participate in survey-based research, we have used conceptual models from this body of research to develop the recruitment approaches for the proposed research [13]. Incentive Approach First, the opportunity cost [13] approach argues that the typical invitee to the CGN is rational, in the sense that they compare the perceived costs and benefits of participation in a survey. The perceived costs of participating are likely based on the time needed to complete the task, other competing tasks being neglected or postponed, cognitive burden required to comply with the survey requests, the future obligations that participation may incur, and the possible embarrassment or discomfort associated with providing (or not being able to provide) a family medical history. The perceived benefits may include the novelty of participating in a research project, and the possibility that participation will improve the diagnosis, treatment, and prevention of cancer. In general, when perceived benefits outweigh perceived costs, an invitee is more likely to enroll and complete the questionnaires. Providing an enrollment incentive to probands may increase perceptions of the benefits of CGN participation in relation to perceptions of the costs (i.e. time) of participating. The theory of social exchange (SE) [14, 15] argues that the key element affecting participation in survey research is the perceived equity between a person and the institution conducting the study.

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This theory predicts that an individual will enter into and stay within a relationship so long as there is reciprocation and the social ‘ledger’ is roughly in balance. The commodities involved in a relationship include those that are social (trust, approval) as well as economic (money, information) in nature. SE theory may apply to a wide range of obligations and expectations over an extended period of time between individuals and social institutions. The theory is best suited for studying long-term associations between individuals or between individuals and institutions (e.g. spouse or employee-employer dyads). In the case of the CGN, where the individual is being asked to agree to a potentially long-term relationship with the CGN, the ideas embodied in SE theory are relevant. As individuals evaluate whether they will participate, they will likely consider what it means to provide consent to join the CGN in terms of future studies and the potential involvement of family members. Whether there is perceived equity between what the individual will provide and what the individual will receive will depend in part on how well the CGN is able to describe the protocol and persuade individuals of the value of their participation. This, however, is a moot point for persons who are not interested enough to even read about the protocol and the study design. An incentive, as proposed here, will likely increase the chances that the subject will sufficiently consider enrolling in the CGN. According to SE theory, the phone card incentive proposed here additionally will create an inequity such that the individual will feel more obliged to participate. The provision of phone cards is also a well-tailored intervention in that it allows individuals to remain in contact with family members with whom they also have a long-term relationship. Finally, the theory of social isolation suggests that persons who are isolated or alienated from larger society will be less likely to accept an invitation to participate in a study to the extent that the sponsors of the study are viewed as representing the interests of the larger study (i.e. the federal government, the medical profession). This argument has been used to explain lower participation rates among ethnic minorities, persons of low socioeconomic status and the elderly [13]. To some extent, participating in a survey is similar to voting, where more isolated or disenfranchised persons are less prone to involve themselves in an activity that is beneficial to larger society. On the other hand, an invitation to participate can also be seen as an opportunity for the individual to provide input about an issue that can be empowering, thereby reducing his/her level of perceived alienation. It is also possible that the individual will not see the interests of medicine or the government being served but rather the individual’s own family. This attitude may arise if individuals are viewing themselves as cancer survivors or members of high-risk cancer families. An incentive to participate may be especially effective at enhancing cooperation rates among the poor or the most disenfranchised who may view it as a form of appreciation on the part of the CGN. Culturally Appropriate (Pan-Asian) Message Letter The culturally appropriate message letter was conceived based on evidence that individuals from minority cultures report feeling left out or otherwise disenfranchised from research projects and studies [1]. Recruiting Asian individuals poses additional complications of diverse cultures, languages, and perspectives within the label ‘Asian’. Many studies hire staff and provide materials that reflect the language, images, and skin color of the in-

236

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dividuals to be recruited to demonstrate inclusion in all areas of the project. Recruitment through a cancer registry is a commonly used and efficient population-based method of identifying cancer patients for participation in a wide variety of studies. However, SEER-based recruitment offers no option for personal contact prior to recruitment and no option for determining the specific Asian culture of the potential recipient. Therefore, a simple, culturally appropriate message letter was designed that welcomes people to participate in six key Asian languages and in English. The letter is intended to convey a welcoming feeling to the potential participant, so that when the approach packet arrives, or an interviewer calls to determine eligibility and interest, the participant is more likely to engage the interviewer and to ultimately join the CGN. Study Population The Institutional Review Board at both institutions approved the study. Potential participants were identified through the Cancer Surveillance System at FHCRC which collects data on malignancies in thirteen counties in northwestern Washington State and the Cancer Surveillance Program of Orange, San Diego and Imperial County at UCI. Cancer cases are reported mandatorily via state-wide tumor registries; therefore, the majority of cases are identified without patient consent. Participant consent is obtained if a patient chooses to enroll in a study. In this study, Asian men and women age 18 and older, diagnosed with breast, lung, stomach, ovarian, prostate, or colorectal (UCI only) cancer between the years of 1998 and 2002 were eligible if they were able to speak English (FHCRC only) or English or Vietnamese (UCI only). Although beyond the scope of this study to specifically distinguish Asian subgroup differences, it was hypothesized that the Asian community at FHCRC would be composed of multiple ethnicities, including Vietnamese, Chinese, Korean, Japanese, and Filipino, many of whom would had lived in the US longer than the relatively recent immigrant Vietnamese community in southern California, which comprised the largest Asian subgroup for the UCI sample. Study Design Eligible subjects were stratified by age (!50, 650 years) and gender. Potential participants were randomly assigned, through the process of allocation concealment [16], which is used to conceal the randomization assignment until after the randomization process is over, to receive one of the following recruitment approaches: (1) Traditional approach packet: This includes an institutionspecific invitation letter, study brochure (FHCRC subjects) or cancer registry brochure (UCI subjects), consent form, study questionnaire, and business reply envelope. Usual telephone follow-up occurred approximately 1 week after mailing. (2) Traditional approach packet + phone card incentive with mailing: This included the traditional approach packet materials outlined above, with the addition of a phone card incentive printed in English, but with international calling capacity. The incentive, a USD 20 (maximum value) telephone calling card, was included in the approach packet and was highlighted within the body of the invitation letter. (3) Traditional approach packet + pan-Asian message greeting with mailing: This included the traditional approach packet materials outlined above (1), with the addition of a pan-Asian

Wenzel /Bowen /Habbal /Leighton /Vu / Anton-Culver

600 8.8%

Participants

500 400 300 200 100 0

2.6% Jul Sep Nov Jan Mar May Jul Sep Nov Jan Mar May Jul 02 02 02 03 03 03 03 03 03 04 04 04 04

Fig. 1. Example of pan-Asian message letter.

Fig. 2. Percent of Asian participants of total CGN participants as

of July 2004 data transmission (FHCRC and UCI).

message greeting (fig. 1). The colorful cultural message letter says ‘hello’ in Chinese, Tagalog, Vietnamese, Japanese, and Korean. Each letter was personalized to include the subject’s name on the left. The message welcomes the subject to the Cancer Genetics Network and contains the phrase ‘Diversity makes the Network stronger, a member of our team will be in touch with you soon’. This sheet was placed first (on top) in the packet of materials. (4) Traditional approach packet + pan-Asian message greeting + phone card incentive with mailing: This included the traditional approach packet materials outlined above, in addition to the phone card incentive and personalized pan-Asian message greeting (1–3). At UCI, the study introductory letter, consent form, and questionnaire were translated into Vietnamese. Subjects who were identified as Vietnamese in the Cancer Registry received a mailout packet containing study documents in both English and Vietnamese. Telephone follow-up with identified Vietnamese subjects was conducted with a bilingual Vietnamese interviewer; an English-speaking interviewer followed up with other Asian participants. Measures Core Data Elements Survey Participants were asked to complete the standard CGN Core Data Elements (CDE) Survey. The CDE is an extensive questionnaire that evaluates the participant’s demographic information (age, education level, marital status, and ethnicity), general medical history (i.e. personal cancer history, surgeries received), and cancer family history. In terms of cancer family history, participants were asked to provide information (i.e. relative’s name, type of cancer, age at diagnosis, current age, and vital status) about first-, second- and sometimes third-degree relatives from both maternal and paternal sides of the family. The CDE Survey took approximately 30–40 min to complete.

Minority Recruitment within Asian Populations

Refuser Survey Participants who declined enrollment in the CGN were asked to complete the refuser survey over the phone. The refuser survey consists of four questions that address patient’s ethnicity, language(s) spoken at home, reasons for disinterest in CGN enrollment, and effect, if any, the approach materials had on their decision to participate. The purpose of the survey is to evaluate the effectiveness of our randomized trial and to identify potential CGN enrollment barriers. The ethnicity, language, and incentive questions were also included in the core survey. Data Analysis The overall response rate in the CGN population-based centers ranges from 60 to 75%. However, the response rate calculation was based on a primarily Caucasian sample. We anticipated that the response rate for an Asian sample would be less than the response rate for Caucasians, by at least 20%, based on previous research [1]. Therefore, based upon the experience of this investigative team in conducting other interview-based case-control studies of cancer in the same population, approximately 40% of the eligible cases were projected to return completed questionnaires. Since the main outcome of this study was to enroll Asian participants into the CGN, the change in Asian enrollment due to the intervention was measured. Outcome evaluations of participant enrollment in the CGN were conducted following the study invitation. Specifically, CGN records at both centers were reviewed to determine whether eligible subjects provided informed consent to participate in the CGN and completed the CDE survey. Descriptive statistics were generated to characterize the study sample. Comparisons between participation rates were conducted using the likelihood ratio ␹2 test. Logistic regressions were used to estimate odds ratios and 95% confidence intervals, where participant enrollment (accept or decline) in the CGN was the outcome of interest. These models were based on all individuals invited to join the CGN. Data were analyzed with SAS for Windows, v8.2 [17].

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Identified cases via SEER cancer registry (n = 1,096) randomization

Fig. 3. Study disposition of identified sub-

Traditional intro letter (n = 280)

USD20 phone card + intro letter (n = 278)

Message letter + intro letter (n = 278)

USD20 phone card + message and intro letter (n = 260)

Interviewed (20%)

Interviewed (20%)

Interviewed (28%)

Interviewed (25%)

Declined (28%)

Declined (27%)

Declined (25%)

Declined (32%)

Dead/ no contact (51%)

Dead/ no contact (53%)

Dead/ no contact (47%)

Dead/ no contact (43%)

jects.

Results

Primary Outcome: Overall Enrollment The primary objective was to increase Asian participation into the CGN. As shown in figure 2, a 6.2% gain in Asian participants over a 2-year period at FHCRC and UCI was achieved. The total composition of the CGN at FHCRC and UCI prior to targeting Asians for enrollment was only 2.6%. By the end of this study, almost 9% of enrollees at the two centers were Asian. This contributed to a 2% CGN-wide increase in overall Asian enrollment. The overall participation rate (after excluding deceased and no contact subjects) was 45.5%. Figure 3 describes the disposition of all identified study subjects, as recommended by Altman et al. [18]. In this, 1,096 subjects were identified for this study at FHCRC and UCI, 280 subjects were randomized to receive the standard mailing, 278 received the standard mailing plus phone card, 278 received the standard plus message letter, and 260 received the standard packet plus phone card and message letter. A total of 256 subjects enrolled into the CGN by completing the questionnaire: 57 (20%) in the traditional mail-out group, 56 (20%) in the phone card group, 77 (28%) in the message letter group, and 66 (25%) in the combined group. A total of 307 subjects declined to participate in the CGN: 79 (28%) in the traditional mail-out group, 75 (27%) in the phone card group, 70 (25%) in the message letter group, and 83 (32%) in the combined group. The major reasons cited for declining included no interest, bad health, and time com238

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mitment involved in participating in the CGN. Finally, 533 people were not available to study due to ineligibility, inability to be contacted due to wrong address and phone number, or death: 144 (51%) in the traditional mail-out group, 147 (53%) in the phone card group, 131 (47%) in the message letter group, and 111 (43%) in the combined incentive group. Overall participation rates did not differ among study arms with data from both centers combined: 41.9% for controls (i.e. traditional recruitment arm 1), 42.8% for the phone card group, 52.4% for the message letter group, and 44.3% for the combined group. Demographic and Medical Variables Table 1 provides a description of the study participants’ demographics. Participants were 34.8% Vietnamese, 17.6% Japanese, 14.8% Filipino, 13.3% Chinese, 4.3% Korean, and 15.2% other Asian ethnicities. Additionally, 54.7% of participants were female, 78.5% were age 50 or more, and 61.3% completed some college or more. The majority of participants were diagnosed with breast or prostate cancer. Other diagnoses included colorectal, lung, liver, cervix, ovarian, and stomach cancers. Participation by Center With the control group set as the reference, subjects were as likely to participate in the study whether they received an incentive or not (table 2). However, a logistic regression by center revealed that the FHCRC group did have a significant effect of incentives on enrollment. Specifically, the USD 20 phone card group was almost 3 times Wenzel /Bowen /Habbal /Leighton /Vu / Anton-Culver

Table 1. Demographics of enrolled participants (n = 256)

n

Table 2. Odds ratio (OR) and confidence interval (CI) that a subject will participate in the CGN, by study arm

% Group

Ethnicity Vietnamese Japanese Filipino Chinese Korean Other Sex Male Female Age at interview <50 years ≥50 years Education High school or less Some college or more Unknown

89 45 38 34 11 39

34.8 17.6 14.8 13.3 4.3 15.2

116 140

45.3 54.7

55 201

21.5 78.5

96 157 3

37.5 61.3 1.2

more likely to enroll than the control group; participants who received the message letter were 4.5 times more likely to enroll, and the two-incentive group (phone card plus message letter) was 3 times more likely to enroll. Although an incentive effect was not found at UCI, it is important to note that Vietnamese participants were significantly more likely to enroll in the CGN than other Asians, independent of gender and age (p ! 0.0001) (49 vs. 29%). This effect held for each study arm, although it was statistically significant in the traditional study arm only (p = 0.0028; table 3). Specific to family history, no significant difference was observed in participation rates between those with no first-degree relatives with cancer and those with at least one first-degree relative with cancer. Factors Influencing Decision to Enroll in the CGN Various factors were cited as being influential to participants in deciding to enroll in the CGN. Chief among them was the ability to help find new treatments for cancer (91.4%), help find new genes that may cause cancer (89.3%), help their children and family (84.8%), help learn more about hereditary cancers (72.5%), and do their part to help science (57.6%).

Discussion

Representation from an ethnically diverse community is an essential component of the CGN. However, barriers to recruitment challenged that goal. Consequently, withMinority Recruitment within Asian Populations

FHCRC and UCI Traditional USD 20 phone card Message letter USD 20 card, message letter FHCRC only Traditional USD 20 phone card Message letter USD 20 card, message letter UCI only Traditional USD 20 phone card Message letter USD 20 card, message letter

Subjects

OR

CI

57 56 77 66

1 1.04 1.53 1.1

0.64–1.68 0.95–2.44 0.69–1.76

15 26 28 30

1 2.89 4.67 3.33

1.02–8.19 1.50–14.53 1.19–9.37

42 30 49 36

1 0.69 1.17 0.74

0.39–1.24 0.68–2.00 0.43–1.29

Table 3. Participation rates (%) by arm and ethnicity

Study arm

Vietnamese (n = 85)

Other Asian (n = 72)

Overall** None* USD 20 phone card Message letter Both

48.9 57.5 42.1 54.3 42.0

28.6 28.4 24.1 35.8 25.0

* p = 0.0028; ** p < 0.0001. Dead/no contact was excluded from analysis, and only UCI data were used.

in the CGN the importance of developing and evaluating novel methods for identification and enrollment of ethnic minority groups in cancer genetics research became a priority [1]. The study reported herein tested registrybased traditional and novel recruitment approaches, resulting in a 6.2% gain in Asian participants over a 2-year period in two of the CGN centers, and a 2% CGN-wide increase in overall Asian enrollment. Although initial analyses suggested that increased participation was not linked to a particular ‘recruitment’ arm, examination of these data by center (FHCRC or UCI) revealed important differences. First, at FHCRC, where all study arms were conducted in English, all novel recruitment arms significantly improved enrollment beyond the traditional method. This suggests that at least Community Genet 2008;11:234–240

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among this population of Asians, who may be more acculturated than those at UCI, an enhanced effort beyond just a letter could be appreciated by potential participants, and fruitful in terms of clinical trial recruitment. Interestingly, the greatest enrollment success was the culturally tailored message greeting, which did not include a financial incentive. In addition, inclusion of both a phone card and the message greeting was no different than the phone card alone, perhaps suggesting that ‘more is not necessarily better’. In contrast, the large Vietnamese population was significantly more likely to enroll than other Asians, regardless of study arm. It is further interesting to note that use of the USD 20 phone card seemed to have the least impact on recruitment among both Vietnamese and other Asians at UCI. Similar to the FHCRC experience, these data suggested that the message greeting in itself could be a powerful motivator, and serve as a culturally appropriate invitation to participate in multicultural studies. Use of the cancer registries for potential study participant identification could be considered a study strength, since a denominator is available within which to estimate disease type, time from diagnosis, ethnicity and some demographic information. Further, it could be considered a strength that two geographically and ethnically diverse

populations could contribute to these study findings, which enhances potential to generalize the overall results. However, several study weaknesses exist. First, true acculturation can not be measured by preferred language (i.e. English vs. Vietnamese). In fact, behavioral measures and inventories which assess internalized cultural values could be viewed as better determinants of acculturation, and these were not included in this study. In support of this statement, anecdotal experiences conveyed by the Vietnamese interviewer, who provided tremendous personal follow-up to each recruitment, suggests that some participants were suspect of the phone cards they received and did not want to use them. Further, between-arm comparisons among ethnicities may not be robust since the study was not sufficiently powered to reflect this. Nevertheless, interesting trends emerged for future consideration. To that end, perhaps the greatest weakness and one most amenable to immediate future research is incorporation of culturally sensitive community/advocacy input at the inception of study development. This could precede a qualitative component to ensure that barriers are addressed to the extent possible. This area of research requires additional focused attention from experts in the cultures and issues of ethnic minority communities in order to enhance cancer genetic study representation among this minority population.

References 1 Hughes C, Peterson SK, Ramirez A, Gallion KJ, McDonald PG, Skinner CS, Bowen D: Minority recruitment in hereditary breast cancer research. Cancer Epidemiol Biomarkers Prev 2004;13:1146–1155. 2 Kagawa-Singer M: Improving the validity and generalizability of studies with underserved U.S. populations: expanding the research paradigm. Ann Epidemiol 2000; 10(8 suppl):92–103. 3 Paterniti DA, Chen MS Jr, Chiechi C, Beckett LA, Horan N, Turrell C, Smith L, Morain C, Montell L, Luis Gonzalez J, Davis S, Lara PN Jr: Asian Americans and cancer clinical trials: a mixed-methods approach to understanding awareness and experience. Cancer 2005;104(suppl 12):3015–3024. 4 Tu SP, Chen H, Chen A, Lim J, May S, Drescher C: Clinical trials: understanding and perceptions of female Chinese-American cancer patients. Cancer 2005; 104(suppl 12):2999–3005. 5 Nguyen TT, Somkin CP, Ma Y: Participation of Asian American women in cancer chemoprevention research: physician perspectives. Cancer 2005;104(suppl 12):3006–3014.

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6 Nguyen TT, Somkin CP, Ma Y, Fung L-C, Nguyen T: Participation of Asian-American women in cancer treatment research: a pilot study. J Natl Cancer Inst Monogr 2005; 35: 102–105. 7 Hussain-Gambles M, Leese B, Atkin K, Brown J, Mason S, Tovey P: Involving South Asian patients in clinical trials. Health Technol Assess 2004; 8:1–109. Review. 8 Liang W, Yuan E, Mandelblatt JS, Pasick RJ: How do older Chinese women view health and cancer screening? Results from focus groups and implications for interventions. Ethn Health 2004;9:283–304. 9 Maxwell AE, Bastani R, Vida P, Warda US: Strategies to recruit and retain older Filipino-American immigrants for a cancer screening study. J Community Health 2005; 30:167–179. 10 Kwok C, Sullivan G: Chinese-Australian women’s beliefs about cancer: implications for health promotion. Cancer Nurs 2006;29: E14–21. 11 Kwok C, Sullivan G: Influence of traditional Chinese beliefs on cancer screening behaviour among Chinese-Australian women. J Adv Nurs 2006;54:691–699.

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12 Anton-Culver H, Ziogas A, Bowen D, Finkelstein D, Griffin C, Hanson J, Isaacs C, Kasten-Sportes C, Mineau G, Nadkarni P, Rimer B, Schildkraut J, Strong L, Weber B, Winn D, Hiatt R, Nayfield S: The Cancer Genetics Network: recruitment results and pilot studies. Community Genet 2003;6:171–177. 13 Groves RM, Couper MP: Nonresponse in Household Interview Surveys. New York, John Wiley & Sons, 1998. 14 Dillman DA: Mail and Telephone Surveys: The Total Design Method. New York, Wiley, 1978. 15 Goyder J: The Silent Minority: Nonrespondents on Sample Surveys. Boulder, Westview, 1987. 16 Schulz KF, Chalmers I, Hayes RJ, Altman DG: Empirical evidence of bias. Dimensions of methodological quality associated with estimates of treatment effects in controlled trials. JAMA 1995;273:408–412. 17 The SAS System for Windows (version 8). Cary, SAS Institute, Inc, 1999. 18 Altman DG, Schulz KF, Moher D, Egger M, Davidoff F, Elbourne D: The revised CONSORT statement for reporting randomized trials: explanation and elaboration. Ann Intern Med 2001;134:663–694.

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Community Genet 2008;11:241–249 DOI: 10.1159/000116878

Strategies and Stakeholders: Minority Recruitment in Cancer Genetics Research Rosalina D. James a Joon-Ho Yu b Nora B. Henrikson b Deborah J. Bowen c Stephanie M. Fullerton a, b for the Health Disparities Working Group a c

Department of Medical History and Ethics and Genome Sciences, b Institute for Public Health Genetics, and Social and Behavioral Sciences, School of Public Health, Boston University, Boston, Mass., USA

Key Words Cancer Genetics Network ⴢ Minority recruitment ⴢ Cancer

Abstract The Cancer Genetics Network (CGN) is one of a growing number of large-scale registries designed to facilitate investigation of genetic and environmental contributions to health and disease. Despite compelling scientific and social justice arguments that recommend diverse participation in biomedical research, members of ethnic minority groups continue to be chronically underrepresented in such projects. The CGN studies reported in this issue used strategies well documented to increase minority participation in research activities, including use of community-targeted materials, addressing community trust concerns, and the adoption of personalized and flexible research protocols. Here, we review the outcome of these efforts to increase minority recruitment to the CGN, and ask what lessons the findings suggest for future minority recruitment initiatives. Copyright © 2008 S. Karger AG, Basel

Members of the Health Disparities Working Group (Center for Genomics and Health Care Equality, University of Washington, Seattle, Wash.) are, in alphabetical order: Llilda Barata, Deborah J. Bowen, Wylie Burke, Leah Ceccarelli, Julia Crouch, Karen L. Edwards (CoChair), Stephanie Malia Fullerton (Co-Chair), Danielle Frank, Kelly Fryer-Edwards, Marisa Gallo, Julie N. Harris, Nora B. Henrikson, Suzanne Holland, Rosalina D. James, Patricia C. Kuszler, Nancy Press, and Joon-Ho Yu.

© 2008 S. Karger AG, Basel 1422–2795/08/0114–0241$24.50/0 Fax +41 61 306 12 34 E-Mail [email protected] www.karger.com

Accessible online at: www.karger.com/cmg

The Cancer Genetics Network (CGN) is just one of a growing number of large-scale registries designed to facilitate investigation of genetic and environmental contributions to health and disease [1]. Unless targeted to members of a single ethnic and/or racial group [2–4], such resources are expected (indeed, mandated) to recruit from minority populations in numbers commensurate with demographic proportions to advance key scientific and societal objectives. In practice, however, facilitating minority participation in biomedical research has been challenging [5–8]. The authors in this issue have attempted to meet that challenge head-on. Here, we review the outcome of efforts to increase minority recruitment to the CGN, and ask what lessons the findings suggest for future minority recruitment initiatives.

Diversity Facilitates Equity in Scientific and Social Advancement

As described by Bowen’s group, there are compelling reasons for the CGN’s interest in identifying methods to improve participation from members of ethnic minority communities. Scientifically, increasing minority participation leads to diverse representation and allows for a comprehensive description of genetic susceptibility to cancer. Minority enrollment allows investigators to compare the spectrum of cancer-associated genetic variants across populations, estimate prevalence of susceptibility variants, and determine relative risks of cancer associatRosalina D. James University of Washington, Medical History and Ethics Box 357120 Seattle, WA 98195-7120 (USA) Tel. +1 206 616 1453, Fax +1 206 616 4978, E-Mail [email protected]

ed with these variants in particular populations [9–11]. A diverse resource also facilitates the prospective assessment of environmental modifiers of genetic susceptibility and translation of genetic discovery to specific programs of screening and therapeutic intervention [12–14]. Key social and behavioral issues, such as potential ethnic differences in response to interventions, are only possible to address with a diverse sample. From a social justice perspective, there are equally important rationales for the emphasis on minority recruitment. While much progress has been made to curb cancer incidence and mortality in the U.S. over the last few decades, these declines have not been equally realized by all populations [6, 15]. The broad knowledge base facilitated by large-scale cancer genetics research is expected to augment downstream access to the benefits of scientific discovery. It is argued that only through minority involvement in CGN registry will downstream studies effectively address and resolve issues of health disparities specific to these populations. For example, population data generated through pharmacogenetic investigations could lead to applications that take into account racial allele frequency with respect to drug response and interactions [16]. Genetic services could also be targeted in a culturally appropriate manner and test specificity could incorporate population-specific data, ensuring that significance of results is not inappropriately generalized across dissimilar populations [10, 15, 17]. These arguments suggest benefits associated with access to research participation are linked to including underrepresented groups in scientific investigation, extension of health gains to the medically underserved, and enhanced awareness of the promise of biomedical advances [15, 18, 19]. In other words, achieving full participation of ethnic and/or racial minorities is not only desirable, it is a necessary prerequisite for the CGN achieving its fullest potential to provide knowledge that impacts cancer for all populations in the U.S.

If Minority Recruitment Is So Important, Why Is It So Hard?

Despite the many reasons to recommend diverse participation in biomedical research, the initial underrecruitment of minority participants to the CGN [Bowen and colleagues] is typical of enrollment experience for such projects [20–22]. Several major barriers are commonly recognized as contributing to the problem of low minority recruitment. First, significant negative histori242

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cal events have led to a pervasive distrust of research and researchers. Notorious human subjects violations in the conduct of research with specific minority communities, such as the Tuskegee syphilis experiment [23], are well known and widely repeated, contributing to an overall distrust of biomedical research among African Americans [24]. Unfortunately, such abuses are not a phenomenon of the past, as evidenced by the recent controversy surrounding the misuse of genetic samples from the Havasupai tribe of southern Arizona [25]. In this case, members of a Native American tribe provided blood samples for diabetes-focused studies, a disease that deeply affected this small geographically isolated population. However, over the course of more than a decade samples were shared between researchers and universities to investigate mental disease, inbreeding, and human migratory patterns. With more than twenty scientific articles published and several federal grants awarded via analysis of the samples, two federal lawsuits ensued by Havasupai members, and by the tribe, against involved researchers and Arizona State University claiming misuse of their samples. The dispute now revolves around whether informed consent processes adequately covered the use of blood samples for nondiabetes research [26]. Regardless of legal interpretations of the research consent process, the Tribe maintains that their intentions were for these samples to solely be used for diabetes-related research. As a consequence of these events, the Havasupai have banned all research activities within their jurisdiction and the case has had a chilling effect on academic partnerships with many US-based Native American communities. Such examples illustrate a chasm between standard research practices and community interests in which human subjects misconduct and misunderstandings regarding data use can serve to profoundly undermine trust. Differences in coverage and access to basic services among racial and/or ethnic minorities compared to the general population also contribute to profound inequities in health and healthcare, exacerbating skepticism that research conducted by mainstream academics will ever directly benefit these groups [7, 10, 27, 28]. Put simply, many of the medically underserved in this country seem to have little faith that promises of genetic research will lead to improvement in health care for their own people. In many instances, challenges to overcoming barriers for genetic registry recruitment stem from the need to tackle these more fundamental issues of trust and social inequities within the context of traditional research protocols and time frames. James/Yu/Henrikson/Bowen/Fullerton

CGN Strategies That Influence Minority Participation in Biomedical Research

Several strategies stemming from community-based participatory research have been suggested to increase participation among ethnic and racial minorities in biomedical research that may be relevant to cancer genetic registry recruitment. These methods include provider referral of minority clients, recruiting from cancer registries, community outreach to increase education and awareness, and use of population-targeted materials [29, 30]. Self-referral is another common means of recruiting subjects into cancer genetics studies, but the indirect voluntary component of this method tends to be a barrier for minority participation. One recent study, for example, noted that significantly fewer African American than Caucasian women volunteered to take part in a randomized trial comparing education methods on hereditary breast cancer [30]. For reasons cited above, engaging marginalized populations requires that attention must first be paid towards understanding and addressing competing priorities and concerns that undermine participation in, or fuel outright resistance to, genetic studies among minority groups. A number of frameworks have been articulated for developing research partnerships that can be applied, in part, towards gaining minority community support for registry recruitment efforts [31–34]. These structures emphasize an ethical principle of respect for community through early and consistent consultation. Many strategies for establishing respectful collaborations are rooted in community-based participatory research practices including development of community-representative boards to highlight issues of concern regarding the project, and employment of a facilitator to ensure productive communication between community and research partners. The consultation element is especially crucial when groups possess defined political or review processes such as tribal entities, and negotiations may require a shift of traditional research control to the community in exchange for their approval and support of the partnership [32, 35–37]. Some have suggested creating tangible benefits for community endorsement and collaboration including compensation in the form of resources for health care infrastructure, reimbursement of costs, or even revenues from for-profit ventures or industry-driven research [38]. The CGN studies reported in this issue attempted to enhance minority participation in the registry using some of these well-documented strategies derived from community-based approaches such as Minority Recruitment in Cancer Genetics Research

targeted or tailored materials, attending to trust concerns, and the pursuit of varied and flexible approaches (table 1). Here we highlight some of their findings and discuss directions for future research. Targeting Individuals with Preliminary Exposure to Cancer Genetics Targeting high-risk cancer patients and families enriches the pool of potential subjects with a foundation of experience, knowledge, fears, and questions concerning genetic disease [30, 39]. In this issue, Skinner and colleagues experienced high overall CGN participation (75%) among African Americans recruited from tumor registries and oncology clinics, regardless of targeted materials. The study sample had high levels of trust and education, and perceived few barriers to genetic research. Recruitment of individuals of low education and socioeconomic status, however, presents a multitude of practical and social barriers that often require more personalized interventions to increase participation in genetic registries [30, 40]. Use of Targeted Materials Linguistically or culturally relevant materials facilitate awareness of the benefits of research participation in audiences that may not otherwise absorb generic messages, as was observed by Wenzel and colleagues. Investigators examined the relative efficacy of four mail-out recruitment methods conducted through University of California Irvine and Fred Hutchinson Cancer Research Center located in Washington State. Potential subjects were randomly assigned to receive a traditional packet containing basic study materials or one of three packets including the traditional materials plus an incentive, a personalized invitation with Pan-Asian language greetings, or a combination of the incentive and tailored message. All mailings were followed up with a telephone call, but the California study translated packet information and provided follow-up in Vietnamese. The fact that the greatest proportion of new recruits were of Vietnamese ethnicity suggests removal of language barriers alone can dramatically influence active participation for this population. This point was demonstrated in another study where a video narrated in Spanish was used to recruit non-English speaking women to a research project on home safety visits. In this example, barriers of language and low literacy among Hispanic mothers were addressed through visual, language tailored, communication tools [41]. This finding raises the question of which subgroups may be predisCommunity Genet 2008;11:241–249

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Table 1. Results of CGN minority recruitment intervention strategies Authors

Sample

Intervention strategies

Results

Lessons learned

strategic targeted/ goal community flexible recruit- tailored concor- representa- methment materials dance tive staff ods

Olsen 1,283 African Americans and from 4 state chapters; colleagues sorority members with genetic awareness

!

!

!

Patterson African Americans from and Texas; recruited from colleagues breast cancer clinics and tumor registries

!

!

!

Ramirez 458 Hispanics from Texas; ! and recruited from cancer colleagues registries with personal or family cancer history

28 (2.3%) enrolled in CGN: Recruitment cost high per enrollee; 11% recruited through presentation targeted direct mail campaign more at regional meetings; effective than educational session 89% recruited via mailings !

!

233 enrolled in CGN: 66% via hospital-based sources; 28% from churches/recreation facilities; 6% from social/religious networks; mainly recruited from cancer clinics and family gatherings rather than counseling sessions; face-to-face recruitment was primary strategy employed (vs. self-referral for Caucasians)

Racial concordance of recruiter and target population, and cultural competence of recruiter important; 1 year targeted recruitment exceeded prior enrollment over 6 years; flexibility of staff schedules necessary to accommodate prospective participants; measures of intermediate steps valuable for intervention development

!

!

!

153 (33%) enrolled in CGN: 42% via standard mailing, 28% using tailored materials, and 30% with telephone follow-up; tailored magazine was viewed favorably; rating for follow-up call low; refusal surveys indicated low interest or no time

Flexibility of research staff required to complete follow-up calls; without call support, tailored materials had little impact; history of cancer did not predict participation; consent difficult; cost of implementing these methods should be researched

Skinner 545 African Americans ! and from North Carolina and colleagues Washington, DC; recruited through oncology clinics (DC) and tumor registries (NC)

!

!

412 (75%) enrolled in CGN: 68% from targeted and 68% from standard brochures; trust and perceived barriers associated with enrollment

Targeted brochures had no effect on attitudes towards CGN or enrollment; invitation to participate may have been enough to recruit from this study sample

Wenzel 563 Asians from California and and Washington; English or colleagues Vietnamese speakers; cancer patients

!

!

256 (45%) enrolled in CGN: 20% via traditional mailing; 20% with phone card incentive; 28% with tailored message letter; and 25% with combined methods; achieved 6.2% gain in Asian participation over 2 years

Greatest success with culturally targeted letter; phone card incentive met with suspicion

!

posed to support goals of the CGN provided that communication barriers are removed. Distributing targeted materials alone is often insufficient to increase genetic registry and research participation, thus emphasizing the need to determine when and how to employ additional recruitment strategies. Skinner and colleagues found no improvement in enrollment rates among targeted brochure recipients compared to individuals that received standard brochures. Similarly, although a culturally-relevant magazine was well received in the study by Ramirez and colleagues, it played no detectable role in boosting actual Hispanic CGN enroll244

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ment. Additional examples suggest that providing information designed with respect to a particular target group does not always translate into higher participation rates [42–44]. Culturally appropriate product development involves community input and specialized materials for each target population, which translates into increased project expenses. The current scarcity of research funding implies that more work needs to be done not only to determine which groups are likely to respond to targeted materials, but also what motivations drive their choice to enroll. Tackling language and communication factors are clearly important, but identifying when additional meaJames/Yu/Henrikson/Bowen/Fullerton

sures are necessary to address underlying issues of trust is critical for researchers striving to make the most impact with limited resources. Acknowledging and Addressing Issues of Trust Goal concordance between research teams and their partners is a way to build trust at the group level and can facilitate access to study subjects [27, 31, 36, 45]. Olsen’s group, for example, made a concerted effort to match the research team’s goal of recruitment into genetic registries with those of the community partner. The stepwise collaboration with Zeta Phi Beta allowed investigators to test intervention methods among minority women who had previous exposure to genetic education. While the total enrollment yield of the study was just 3%, that margin might have been enhanced by incorporating additional individual interaction rather than relying on group presentations and mailings [30, 40]. Nonetheless, by achieving project endorsement, the research team successfully gained access to a large number of subjects using a theme directly aligned with existing sorority interests. Involving community members as consultants and intervention staff also builds trust at both the community and individual levels [28, 40, 44, 46, 47]. Patterson and colleagues employed an African American nurse on-site in high-risk clinics and cancer treatment centers to coordinate study efforts and maintain visibility among patients and staff. The consistent presence of such a highly trained, trusted individual can be instrumental in breaking down a number of important obstacles to project acceptance and retention [28, 40, 42, 44, 46, 47]. Members embedded in the community setting can contribute wisdom regarding the usual customs, practices, taboos, and incentives that may be weighed against distrust and cultural differences placed on the value of contributing to scientific knowledge [24, 28, 40]. Flexibility of Intervention Methods and Evaluation Measures Intervention methods and evaluation measures that incorporate room for adaptation to community input and issues have potential to maximize minority participation in research [30, 31, 39, 44, 48, 49]. Many studies have shown that factors such as lack of time, competing obligations or needs, or access issues can deter potential subjects who would otherwise be interested in research or clinical studies [10, 39, 50]. Flexibility within the protocol is often required to successfully deliver interventions in hard-to-reach populations. Adapting methods to accommodate community or individual needs increases Minority Recruitment in Cancer Genetics Research

research involvement by removing barriers for those who would wish to participate [31, 39, 51]. The use of follow-up phone calls to reduce attrition and establish trust at the individual level has increased participation in clinical trials, health surveys, and genetic research studies [30, 52]. In this issue, the arm of the Ramirez study with the greatest number of new recruits employed bilingual staff that assessed population needs and altered their schedules as necessary to ensure interaction with each study subject (table 1). Such changes included identifying the best time to call people, and keeping records of alternate phone numbers. These methods resulted in increased enrollment relative to the arm that provided materials only. With a multitude of variables contributing to successful minority enrollment in the CGN, evaluating the progressive gains of stepwise interventions makes sense as methods are developed for different target populations. For example, telephone contact with research subjects allowed the Patterson team to gather updated family cancer histories and inquire whether genetic information was shared within African American family units (table 1). This information was used to document elements of secondary study outcomes that shed light on the intervention process, rather than simply reporting end enrollment yield. Data collected on intermediate aspects of an intervention can also be of value to partnering communities with a goal of developing effective methods to encourage members’ participation in health activities [44, 48, 49, 51]. Thus, creative measures and evaluation allow researchers insight into better intervention methods and provide incentives for community collaboration. The fact that relative success was realized when recruitment occurred through oncology clinics or registries suggests that individual choice to enroll may be largely driven by personal connections to cancer. Identifying individuals within communities already motivated by making an impact on cancer is critical for future recruitment protocols to focus energy and limited resources. These audiences may be more likely to find personal benefit in doing their part to alleviate or eradicate this disease. Community support of CGN activities could provide powerful direction for creating awareness and reaching greater numbers of minority individuals that would have heightened motivation for involvement in this type of research. The Olsen study, for example, may have had greater enrollment by enlisting community partners to deliver presentations at the conference. An innovative educational session could be developed that attracted sorority members with cancer experiences, increasing personal interaction among a smaller group of Community Genet 2008;11:241–249

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individuals with awareness of both the disease and genetics. Follow-up interviews with enrollees from the Wenzel mailings might reveal unexpected connections to cancer undetected by personal or familial demographics that could explain their motivation for getting involved in the study once language barriers were removed. Certainly, strategic recruitment from registries and oncology clinics appears to have contributed to enrollment of African Americans and Hispanics by the teams of Patterson, Ramirez, and Skinner. In these cases, targeted or tailored materials may have facilitated understanding about the CGN registry and its goals, which was sometimes enough for individuals to make an informed decision to participate. This result speaks to how promises of genetics and technology may be viewed as hopeful endeavors to those familiar with the devastation associated with cancer diagnosis or risk. Intervention strategies that acknowledge and address important issues surrounding research participation in minority communities can effectively increase enrollment in the CGN. Targeting special populations predisposed to messages of genetic research enhances recruitment yield, and providing targeted materials generates project awareness while overcoming language or cultural obstacles. Taking time and effort to become entrenched in a community setting, and to communicate realistic harms and benefits of genetic research participation, can establish researchers as trustworthy partners. Finally, flexible methods and project evaluation measures allow research teams to collaboratively develop novel interventions to increase minority enrollment in genetic studies. While the tools are available to effectively boost minority participation, such interventions necessitate greater amounts of money, time, flexibility, and active outreach on the part of the research project. To accomplish these objectives, researchers must successfully align their own interests with those of minority communities and research funders.

Moving Forward

The CGN minority enrollment studies reviewed above suggest that strategies aimed at increasing recruitment have strong potential for success, given appropriate time and resources. Adequate levels of minority enrollment will require additional research and refinement of these strategies and a more detailed and critical evaluation of the criteria used to measure success. Investment of sufficient time and financial resources will be important for 246

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learning how best to go about recruitment. Furthermore, the interests of various stakeholders will need to be balanced through collaborative endeavors to address which health concerns are investigated, and what constitutes benefits or harms for partnering parties. Time and Resources Enable Relationship Building The literature repeatedly suggests that strong relationships are important for eventually improving minority enrollment, but funding mechanisms and academic pressures limit resources that can be devoted to this process [44, 49]. Faced with the challenge of meeting financial and numerical recruitment bottom lines, researchers have tended to turn to culturally tailored pamphlets or concordant intervention staff as feasible strategies that fit the constraints of standard grant structures. As was observed by several studies in this issue, multiple community-based participatory research-based strategies tailored to the needs and interests of each target population are necessary to realize significant improvement in minority recruitment levels. Moreover, methods responsive to community input can maximize productive relations if federal funding guidelines are expanded to accommodate, finance, and reward researchers that incorporate these approaches into grant proposals [44]. Researchers can motivate and guide funders to encourage sound community-based research practices by asserting the relevance and significance of such strategies in their requests for support. Time to establish community relations, flexibility of project boundaries, and support for sharing benefits of discovery must be built into the overall structure of research plans. Given opportunities to interact with prospective research partners, communities may help identify how projects could be designed to include satisfactory benefits and minimize research harms [38]. Researchers must seek opportunities to attend community events on local turf without the pressure to sell a predetermined proposal, thus allowing investigators to plan relevant interventions while establishing a presence in minority communities. Research budgets and timelines should be expanded to fund efforts that include multiple approaches and more personal or face-to-face interaction with minority communities and individuals. Community knowledge is too often a resource that goes unrecognized and untapped, yet has potential to enhance rigorous scientific research that can be translated into direct applications. Many minority groups who have experienced research done on, or about, them with little more involvement James/Yu/Henrikson/Bowen/Fullerton

than providing a letter of support, may now demand that investigators outline what participation will allow them to ‘walk away with’ [31, 36, 53]. This is especially true in the arena of genetic research, where benefits to individuals or communities can be vague, and more immediate concerns of basic healthcare issues can take precedence. Although the appropriate period needed to establish working partnerships varies between minority groups, significant time and human resources for relationship development should be supported as a preliminary phase prior to project start, and as a closure phase that encourages researchers to share data with communities [31, 36]. Aligning Researcher Interests with Those of Communities and Funding Agencies Researchers and racial and/or ethnic minority communities often face different needs and pressures when engaging in the research enterprise. For instance, NIHfunded research requires production of tangible study results within 3-year funding cycles, while outreach to some minority communities can take a full year to build relations before any project planning begins. Community health priorities stem from local needs assessment and may differ from those of researchers whose ideas are influenced by topics relevant to funding sources. Some minority communities may, for example, identify investigating exposure to environmental hazards as a priority over genetic susceptibility research. Developing medical infrastructure for prevention and basic health services is often considered more important than understanding the genetics of cancer. Nonparticipation in genetic activities can be overcome by using established intervention methods that promote open collaboration with minority groups and respectful regard for their role in the research process. Expanding measures of recruitment success to capture intermediary steps of relationship building would provide researchers with incentive to invest time into these phases, while satisfying federal funding requirements. Establishing institutional commitments and gauging the interest of prospective participants by recording attendance at information gatherings, for instance, would serve to support the development of good practice in minority recruitment [48, 51]. Concomitantly, receiving professional ‘credit’ for such achievements through academic measures such as promotions and tenure would stimulate adoption of minority community outreach strategies among investigators, and eventually yield improvements in the bottom line. Minority Recruitment in Cancer Genetics Research

Conclusions

Minority participation in large-scale genetic resources such as the CGN, though challenging, is both achievable and incredibly important. The research described in this special issue of Community Genetics demonstrates a collection of strategies with the range and power to facilitate diverse participation. Genetic susceptibility and its implications for treatment and prevention can matter to prospective minority research participants. One study found that African Americans involved in genetic variant research on smoking susceptibility cited future use of genetic information to improve cessation programs as a reason for participation [54]. Approaches which target those predisposed to participation provide culturally tailored and accessible materials, foster trustworthiness, and employ flexible methods, and evaluation measures can be applied to maximize involvement of racial and/or ethnic minorities and other marginalized groups. Focusing on community strengths rather than just negative health issues may also be a more attractive approach for minority groups who have been inundated with statistics about problems and are motivated by messages of hope [36]. Minority individuals and communities must be invited to collaborate in the research process to achieve representative involvement in genetic discovery. A key component of establishing a trustworthy reputation in any community is open and transparent communication regarding the likely benefits or risks involved in research cooperation [36, 44, 55]. The intended fruits of genetic research imply immediate or future improvement of disease control, but these benefits often take time to come about and direct access to scientific advances may not be available to all individuals. Likewise, scientists must go to great lengths to communicate potential harms, including unintended consequences, of research participation. Open discussion of these topics reflects well on the integrity of the research team and provides minority groups and prospective subjects with the tools to make informed decisions regarding whether benefits outweigh risks. Investigators who acknowledge and respect the impact of historical and current issues in communities can effectively navigate relations between minority groups and government-funded biomedical research [27, 56]. To achieve the desired improvements, time, care, resources, and the foresight and patience of key stakeholders will all be required.

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Author Index Vol. 11, No. 4, 2008

Anton-Culver, H. 234 Banerji, P. 208 Bowen, D.J. 189, 191, 234, 241 Calingaert, B. 224 Chalela, P. 215 Crankshaw, S.S. 224 Davis, H. 208 Domchek, S. 193 Fish, L. 224 Fullerton, S.M. 241 Gallion, K. 215 García Arámburo, S. 215 Griffin, C.A. 201 Habbal, R. 234 Halbert, C.H. 193 Health Disparities Working Group 241 Henrikson, N.B. 241 Hoyo, C. 224 James, R.D. 241 Jasper, C. 224 Jenkins, I.L. 201 Kasten-Sportes, C. 191 Kessler, L. 193

Leighton, N. 234 Malvern, K.T. 201 May, B.J. 201 McCoy, J. 208 Miller, A.R. 215 Olsen, S.J. 201 Patterson, A.R. 208 Penchaszadeh, V.B. 189 Ramirez, A.G. 215 Rao, S.K. 208 Reid, L. 224 Robinson, L.D. 208 San Miguel de Majors, S. 215 Schildkraut, J.M. 224 Shelby, K. 208 Skinner, C.S. 224 Stopfer, J. 193 Susswein, L. 224 Tomlinson, G.E. 208 Vu, T. 191, 234 Wenzel, L. 234 Yu, J.-H. 241

Subject Index Vol. 11, No. 4, 2008

African American(s) 201, 208, 224 – – women 193 BRCA1/2 mutation 193 Cancer 201, 215, 241 – genetic(s) 234 – – registry 224 – – studies 208 Cancer Genetics Network 201, 215, 224, 234, 241

© 2008 S. Karger AG, Basel Fax +41 61 306 12 34 E-Mail [email protected] www.karger.com

Accessible online at: www.karger.com/cmg

Genetics 201 Hereditary breast cancer 208 Hispanics 215 Minority recruitment 208, 234, 241 Recruitment 201, 215 Registry 215 Research 201 Risk perception 193