Development and validation of a stakeholder-driven, self-contained electronic informed consent platform for trio-based genomic research studies

Abstract

Increasingly long and complex informed consents have yielded studies demonstrating comparatively low participant comprehension and satisfaction with traditional face-to-face approaches. In parallel, interest in electronic consents for clinical and research genomics has steadily increased, yet limited data are available for trio-based genomic discovery studies. We describe the design, development, implementation, and validation of an electronic iConsent application for trio-based genomic research deployed to support genomic studies of cerebral palsy. iConsent development incorporated stakeholder perspectives including researchers, patient advocates, institutional review board members, and genomic data-sharing considerations. The iConsent platform integrated principles derived from prior electronic consenting research and elements of multimedia learning theory. Participant comprehension was assessed in an interactive teachback format. The iConsent application achieved nine of ten proposed desiderata for effective patient-focused electronic consenting for genomic research. Overall, participants demonstrated high comprehension and retention of key human subjects' considerations. Enrollees reported high levels of satisfaction with the iConsent, and we found that participant comprehension, iConsent clarity, privacy protections, and study goal explanations were associated with overall satisfaction. Although opportunities exist to optimize iConsent, we show that such an approach is feasible, can satisfy multiple stakeholder requirements, and can realize high participant satisfaction and comprehension while increasing study reach.

Figure 1.

Overall Comparison of Initial (Comprehension) and Follow-up (Retention) Scores

Figure 2.

Overall Participant Satisfaction and Responses for Key Elements Impacting Satisfaction. Darker shades represent higher degrees of Satisfaction

Figure 3.

Association Between Study Elements and Overall Satisfaction Scores a Satisfaction score was scored from strongly disagree (−2) to strongly agree (2) on the survey question “I had an easy time signing up for the Genetic Causes of CP study.” b Comprehension score was defined as the number of comprehension questions answered correctly on the initial survey out of 5 possible. c Retention score was defined as the number of comprehension questions answered correctly on the follow-up survey. d Clarity was scored from strongly disagree (−2) to strongly agree (2) on the survey question “When I joined the study, I understood what I had to do to sign up.” e Adequacy of Information was scored from not nearly enough (−2) to way too much (2) based on the statement “The amount of information I was given about the study was _____.” f Privacy was scored as yes (1) or no (0) on the survey question “Are the privacy protections for the study strong enough?” g Study goal was scored from strongly disagree (−2) to strongly agree (2) on the survey question “When I signed up for the study, I understood that the researchers are trying to find changes in my family’s DNA that cause cerebral palsy.” h Participant trust score was defined as the number of H-TBR scale questions answered as indicating trust. Answers indicating trust are as follows: “Doctors who do medical research care only about what is best for each patient.” (Agree) “Doctors tell their patients everything they need to know about being in a research study.” (Agree) “Medical researchers treat people like “guinea pigs.” (Disagree) “I completely trust doctors who do medical research.” (Agree)