Chapter Two - Stakeholder Views on Returning Research Results
Introduction
Since the completion of the Human Genome Project, vast amounts of genetic and genomic information have been produced from thousands of consenting research participants. Array-based and sequencing technologies have improved and become cheaper and easier to do, resulting in widespread use of these technologies and subsequent generation of large datasets (Mardis, 2011). Alongside advances in genome technologies, the culture and practice of clinical research has become more patient-centered (Kaye et al., 2012), and participants have become more engaged in the research enterprise (Phipps et al., 1999, Psillidis et al., 1997), playing a greater role in establishing biorepositories and collaborating with researchers (Landy et al., 2012, Terry et al., 2007). Additionally, in the clinical setting, the patient–provider relationship is facing changes with the implementation of electronic medical records and convenient patient access to their health information. With the coevolving research dynamics and changes in genomic and informational technologies, it should serve as no surprise that debates have ensued about research participants' right to access their individual data and researchers' duty to return research results. The public's interest in genetics and genomics and associated ethical, legal, and social issues further contribute to this ongoing debate, complicating development of a consensus policy. In this chapter, we will explore the views of key stakeholders (researchers, institutional review boards, and research participants) on this controversial issue and related issues such as informed consent, communication of research results, and cost. This inherently complex issue will continue to pose major challenges for how best to resolve; however, it appears that a consensus is emerging between the stakeholder groups on returning some types of results and now is the time for every researcher to address this issue in their study's protocols and consent documents.
Section snippets
What is a Research Result?
For more than a decade, the issue of returning research results, particularly in genetics research, has been the topic of great debate. Before considering the issue in depth, it is important to first clarify what is meant by “return of results” (Knoppers & Dam, 2011). Returning research results can refer to at least three types of results: (1) a summary of the study's findings that does not include any personal data; (2) an individual research result for a participant that would be anticipated,
Guidelines on Returning Research Results
Several guidelines have been developed with recommended criteria for when to return research results, for any type of study, or for genetic studies specifically and reviewed in depth in several papers (Beskow et al., 2001, Bookman et al., 2006, Canadian Institute of Health Research, 2012, Cassa et al., 2012, Dressler, 2009, Knoppers et al., 2006, National Bioethics Advisory Commission, 1999, National Heart, Lung and Blood Institute, 2004, National Human Genome Research Institute, 2010, National
Stakeholder Views
Three primary stakeholders in the debate about returning research results are researchers, institutional review boards (IRBs), and participants (or the parent/guardian of a minor). This trifecta represents those who design, carry out, and potentially will communicate research results to participants (researchers); those who govern the conduct of human subjects research (IRBs); and those who consent to participate in research and are ultimately impacted by the research results they receive
Extent and Experience with Returning Research Results
Despite current evidence that research participants would like the option of accessing research results and the growing support of researchers and IRBs, even if of little clinical utility, the practice does not appear to be very common (Table 2.1). Several reasons have been given for not returning results, including time constraints, costs, effort required to recontact participants, lack of national or institutional policies or protocols, limited access to genetic counselors or other clinical
Overall Analysis of Stakeholder Positions
Overall, the issue of returning research results pivots on the ethical and legal obligations. In support of the ethical principles of respect, autonomy, beneficence, and reciprocity, several have declared there is an ethical obligation to return results of clinical significance (Fernandez et al., 2003a, Knoppers et al., 2006, Ravitsky and Wilfond, 2006, Shalowitz and Miller, 2005). However, it is unclear if researchers have an ethical obligation to search for results for clinical significance.
Additional Points to Consider
While much of the debate in the literature has focused on understanding stakeholder perspectives, the practice of returning results is quite complex and involves careful consideration of several factors, including informed consent, communication of test result, provisions for follow-up care, and cost. This is not a complete list but includes some of the commonly debated issues related to returning results.
Moving Forward
In general, the attitudes of many researchers, IRBs, and participants appear to have converged on returning results of clinical significance. However, despite this general consensus, the actual practice appears stymied by a lack of an agreed-upon definition of “clinical significance” and uncertainty about how to actually return results. Given the extent and popularity of genetics and genomics research in the media, it seems inevitable that participants will begin to more frequently request
Acknowledgment
The authors are deeply indebted to the assistance of Ms. Rachel Mills with the preparation of this manuscript.
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