Ethical and practical challenges to studying patients who opt out of large-scale biorepository research

Abstract

Large-scale biorepositories that couple biologic specimens with electronic health records containing documentation of phenotypic expression can accelerate scientific research and discovery. However, differences between those subjects who participate in biorepository-based research and the population from which they are drawn may influence research validity. While an opt-out approach to biorepository-based research enhances inclusiveness, empirical research evaluating voluntariness, risk, and the feasibility of an opt-out approach is sparse, and factors influencing patients' decisions to opt out are understudied. Determining why patients choose to opt out may help to improve voluntariness, however there may be ethical and logistical challenges to studying those who opt out. In this perspective paper, the authors explore what is known about research based on the opt-out model, describe a large-scale biorepository that leverages the opt-out model, and review specific ethical and logistical challenges to bridging the research gaps that remain.

Resisting big data exploitations in public healthcare: free riding or distributive justice?

“…More studies, focusing specifically on patients‐citizens who have opted out, are needed (Rosenbloom et al . ) as well as studies on the public's acceptance of ‘authorisation’ – institutional decision‐making for data releases without informed consent – as a data governance mechanism (Aitken et al . ).…”

“…Most of the literature has focused on public or healthcare professional attitudes to health data sharing, but not on analysing these two groups together, or involving the researchers who are using these datasets. More studies, focusing specifically on patients-citizens who have opted out, are needed (Rosenbloom et al 2013) as well as studies on the public's acceptance of 'authorisation'institutional decision-making for data releases without informed consentas a data governance mechanism (Aitken et al 2016). This research was designed to address these gaps, particularly for national data sharing programmes in healthcare by focusing on three groups involved in care.data: health data researchers, General Practitioners (GPs) and citizens who have opted out from, or have campaigned against the programme.…”

Biorepositories

“…One concern is that obtaining specific consent for each study is administratively difficult, time consuming, and costly. 52 Another concern is that participants may be lost during follow-up or may decline future participation when they are re-contacted, either of which can jeopardize the quantity and quality of future research. 53 These factors have led a number of commentators to argue in favor of alternatives to specific consent for future research use of data and biospecimens.…”

“…BioVu's cumulative opt-out rate of 15% suggests to some commentators that its patients have recognized and exercised their right to opt out of future research. 52 Proponents of presumed consent contend that it not only overcomes the high costs and efforts associated with obtaining informed consent for each research use, it also increases research participation by changing the default from exclusion to inclusion. 52 Critics of presumed consent argue, however, that it does not satisfy the requirements for informed consent, because participants are not told, and do not consent to, the purpose, risks, and benefits of each research use of their data and biospecimens.…”

“…52 Proponents of presumed consent contend that it not only overcomes the high costs and efforts associated with obtaining informed consent for each research use, it also increases research participation by changing the default from exclusion to inclusion. 52 Critics of presumed consent argue, however, that it does not satisfy the requirements for informed consent, because participants are not told, and do not consent to, the purpose, risks, and benefits of each research use of their data and biospecimens. Commentators have also raised concerns about the administrative difficulty of adequately informing all potential participants the opportunity to opt out and the enrollment in research of people who do not wish to participate but fail to opt out.…”