Recommendations for ethical approaches to genotype-driven research recruitment

Abstract

Recruiting research participants based on genetic information generated about them in a prior study is a potentially powerful way to study the functional significance of human genetic variation. However, it also presents significant ethical challenges that, to date, have received only minimal consideration. We convened a multi-disciplinary workshop to discuss key issues relevant to the conduct and oversight of genotype-driven recruitment and to translate those considerations into practical policy recommendations. Workshop participants were invited from around the U.S., and included genomic researchers and study coordinators, research participants, clinicians, bioethics scholars, experts in human research protections, and government representatives. Discussion was directed by experienced facilitators and informed by empirical data collected in a national survey of IRB chairs and in-depth interviews with research participants in studies where genotype-driven recontact occurred. A high degree of consensus was attained on the resulting 7 recommendations, which cover informed consent disclosures and choices, the process for how and by whom participants are recontacted, the disclosure of individual genetic research results, and the importance of tailoring approaches based on specific contextual factors. These recommendations are intended to represent a balanced approach—protecting research participants, yet avoiding overly restrictive policies that hinder advancement on important scientific questions.

Can a decision aid enable informed decisions in neonatal nursery recruitment for a fragile X newborn screening study?

“…34,35 In this study, we show that a carefully designed set of informational materials can improve parents' attention to them and, for those who do read them, the IDM format can result in lower rates of participation, an indication that the decision-making process has been influenced. The fact that only 14% of the mothers read the entire decision aid brochure is sobering but should be tempered by the recognition that parents use multiple sources of information on which to base decisions.…”

Engaging Hmong adults in genomic and pharmacogenomic research: Toward reducing health disparities in genomic knowledge using a community-based participatory research approach

“…Whether to return aggregate results or individual results to genomic study participants is a controversial topic (Beskow et al 2012;Beskow and O'Rourke 2015). Beskow et al stated Bproviding aggregate results is not a substitute for meeting obligations concerning individual result^.…”

The ethics conundrum in Recall by Genotype (RbG) research: Perspectives from birth cohort participants

“…5, 2017; consent and ethical study conduct are often blurred 21 . We have attempted to contextualise for birth cohorts the "central tension" between avoiding the possibility of participant harm by revealing unwanted or misunderstood information and avoiding deception when explaining recruitment into RbG studies 12 . A limited number of studies have attempted to assess the effects of incorporating genetic information in intervention designs 22,23 and some demonstrate the strong social and cultural determinants of those influences in the face of information about genetic susceptibility 24 .…”

“…The seven recommendations that emerged from the consensus workshop about recruitment to studies based on genotype 12 provide a good starting point for other biobanks. All have been followed by ALSPAC: (1) participants be made aware of potential for re-contact; (2) participants have a choice about whether to be re-contacted; (3) re-contact be made by a known person or entity; (4) recruitment be based on the biobank's own processes; (5) thresholds for the return incidental findings be considered differently to the return of genetic information during recruitment; (6) genetic research information offered in the context of RbG recruitment should not leave participants uninformed about the study's purpose; and (7) approaches to RbG recruitment be determined in consultation with ethics committees 12 . In the case of No.6, this must be tempered by the nature of the condition with which the variant is associated.…”

“…Seven broad recommendations for RbG/GDR were developed on the basis of research participant interviews (mostly disease-based biobank participants), a survey with IRB Chairs and a consensus workshop 12 . While useful and important, these recommendations are context specific and do not work for all circumstances 8 .…”