Evaluation of the cost and effectiveness of diverse recruitment methods for a genetic screening study

AbstractPurposeRecruitment of participants from diverse backgrounds is crucial to the generalizability of genetic research, but has proven challenging. We retrospectively evaluated recruitment methods used for a study on return of genetic results.MethodsThe costs of study design, development, and participant enrollment were calculated, and the characteristics of the participants enrolled through the seven recruitment methods were examined.ResultsA total of 1118 participants provided consent, a blood sample, and questionnaire data. The estimated cost across recruitment methods ranged from $579 to $1666 per participant and required a large recruitment team. Recruitment methods using flyers and staff networks were the most cost-efficient and resulted in the highest completion rate. Targeted sampling that emphasized the importance of Latino/a participation, utilization of translated materials, and in-person recruitments contributed to enrolling a demographically diverse sample.ConclusionsAlthough all methods were deployed in the same hospital or neighborhood and shared the same staff, each recruitment method was different in terms of cost and characteristics of the enrolled participants, suggesting the importance of carefully choosing the recruitment methods based on the desired composition of the final study sample. This analysis provides information about the effectiveness and cost of different methods to recruit adults for genetic research.

[1]  K. Malterud Qualitative research: standards, challenges, and guidelines , 2001, The Lancet.

[2]  M. Gammon,et al.  Barriers and strategies for sustained participation of African-American men in cohort studies. , 2003, Ethnicity & disease.

[3]  T. Wadden,et al.  Consent for genetics studies among clinical trial participants: findings from Action for Health in Diabetes (Look AHEAD) , 2006, Clinical trials.

[4]  Cary P. Gross,et al.  Recruiting Vulnerable Populations into Research: A Systematic Review of Recruitment Interventions , 2007, Journal of General Internal Medicine.

[5]  J. Garrett,et al.  Great expectations: views of genetic research participants regarding current and future genetic studies , 2008, Genetics in Medicine.

[6]  J. Weitzel,et al.  Influence of genetic discrimination perceptions and knowledge on cancer genetics referral practice among clinicians , 2008, Genetics in Medicine.

[7]  Tomas Hellström,et al.  Transferability and Naturalistic Generalization: New Generalizability Concepts for Social Science or Old Wine in New Bottles? , 2008 .

[8]  C. Skinner,et al.  Factors Associated with African Americans’ Enrollment in a National Cancer Genetics Registry , 2008, Public Health Genomics.

[9]  Ralph B. D’Agostino,et al.  Consent for genetic research in the Framingham Heart Study , 2010, American journal of medical genetics. Part A.

[10]  I. Spruill,et al.  A systematic review of strategies that increase the recruitment and retention of African American adults in genetic and genomic studies. , 2011, The ABNF journal : official journal of the Association of Black Nursing Faculty in Higher Education, Inc.

[11]  A. King,et al.  Minority recruitment into clinical trials: experimental findings and practical implications. , 2012, Contemporary clinical trials.

[12]  M. R. De La Rosa,et al.  Challenges and strategies in recruiting, interviewing, and retaining recent Latino immigrants in substance abuse and HIV epidemiologic studies. , 2012, The American journal on addictions.

[13]  Robert C. Green,et al.  Managing incidental findings and research results in genomic research involving biobanks and archived data sets , 2012, Genetics in Medicine.

[14]  J. LaFountain Inc. , 2013, American Art.

[15]  S. Vedula,et al.  Cost-effectiveness of health research study participant recruitment strategies: A systematic review , 2014, Clinical trials.

[16]  K. Norris,et al.  A systematic review of barriers and facilitators to minority research participation among African Americans, Latinos, Asian Americans, and Pacific Islanders. , 2014, American journal of public health.

[17]  D. Singer,et al.  Public Interest in Medical Research Participation: Differences by Volunteer Status and Study Type , 2014, Clinical and translational science.

[18]  R Core Team,et al.  R: A language and environment for statistical computing. , 2014 .

[19]  S. Oliveria,et al.  Patient Characteristics and Participation in a Genetic Study , 2014, Journal of Investigative Medicine.

[20]  Avni Santani,et al.  Actionable exomic incidental findings in 6503 participants: challenges of variant classification , 2015, Genome research.

[21]  M. Robson,et al.  Genetic Testing Awareness and Attitudes among Latinos: Exploring Shared Perceptions and Gender-Based Differences , 2015, Public Health Genomics.

[22]  Lisa M. Nicholson,et al.  Recruitment and retention strategies in clinical studies with low-income and minority populations: Progress from 2004-2014. , 2015, Contemporary clinical trials.

[23]  J. Schisler,et al.  Using Community-Based Participatory Research Principles to Develop More Understandable Recruitment and Informed Consent Documents in Genomic Research , 2015, PloS one.

[24]  Marylyn D. Ritchie,et al.  Distribution and clinical impact of functional variants in 50,726 whole-exome sequences from the DiscovEHR study , 2016, Science.

[25]  D. Ledbetter,et al.  The Geisinger MyCode Community Health Initiative: an electronic health record-linked biobank for Precision Medicine research , 2015, Genetics in Medicine.

[26]  L. Biesecker,et al.  Participant use and communication of findings from exome sequencing: A mixed methods study , 2015, Genetics in Medicine.

[27]  S. Fullerton,et al.  Genomics is failing on diversity , 2016, Nature.

[28]  J. O'Brien,et al.  Cost and yield considerations when expanding recruitment for genetic studies: the primary open-angle African American glaucoma genetics study , 2017, BMC Medical Research Methodology.

[29]  Anne Lanceley,et al.  Awareness, knowledge, perceptions, and attitudes towards genetic testing for cancer risk among ethnic minority groups: a systematic review , 2017, BMC Public Health.

[30]  A. Kasarskis,et al.  Psychological and behavioural impact of returning personal results from whole-genome sequencing: the HealthSeq project , 2017, European Journal of Human Genetics.

[31]  Roundtable on Genomics Implementing and Evaluating Genomic Screening Programs in Health Care Systems: Proceedings of a Workshop , 2018 .

[32]  N. Risch,et al.  The Clinical Sequencing Evidence-Generating Research Consortium: Integrating Genomic Sequencing in Diverse and Medically Underserved Populations. , 2018, American journal of human genetics.

[33]  B. Bernhardt,et al.  Why Patients Decline Genomic Sequencing Studies: Experiences from the CSER Consortium , 2018, Journal of Genetic Counseling.

[34]  G. Maupomé,et al.  The VidaSana Study: Recruitment Strategies for Longitudinal Assessment of Egocentric Hispanic Immigrant Networks , 2018, International journal of environmental research and public health.

[35]  Daniel R. Jenkins,et al.  Consent for Genetic Biobanking in a Diverse Multisite CKD Cohort , 2018, Kidney international reports.

[36]  E. Green,et al.  Prioritizing diversity in human genomics research , 2017, Nature Reviews Genetics.

[37]  Patrick B. Ryan,et al.  Decentralized and reproducible geocoding and characterization of community and environmental exposures for multisite studies , 2017, J. Am. Medical Informatics Assoc..

[38]  Robert C. Green,et al.  Harmonizing Clinical Sequencing and Interpretation for the eMERGE III Network. , 2019, American journal of human genetics.