Sharing research findings with research participants has long been considered an ethical imperative, in part as a reflection of respect for persons. Results are typically shared in aggregate, in the form of a published manuscript, lay summary, or letter from investigators. Increasingly, the research community has turned its attention to return of individual-level research results (?individual results?) ? a specific research participant?s own research data, not merely aggregate results. The overarching objective of this supplement project is to identify preferences, perceived risks and benefits, challenges, and obstacles related to returning individual results among multiple stakeholder groups including: caregivers, clinical researchers, clinicians, and sponsors. We will: 1) ascertain stakeholder perspectives on return of individual results through qualitative interviews and 2) evaluate and expand our understanding of themes identified in Aim 1, using surveys in independent samples representative of the identified stakeholder groups. The proposed research activities complement the activities of the parent award and will enable preparation for future return of individual results in the parent study.
In ultra-rare pediatric conditions, disease-related information is scarce outside of the research setting and difficult to obtain, heightening interest in research data. Despite strong anecdotal evidence that caregivers of pediatric research participants desire individual results, guiding principles for clinical researchers faced with navigating the ethical and practical issues have not been explored or developed. As a step towards developing such a framework, we will conduct a multi-stakeholder study of return of results in rare childhood-onset neurological conditions to complement the focus of our parent study on CLN3 disease, a rare pediatric neurodegenerative condition.
