Benefit sharing with participants and the public is an important ethical obligation in biomedical research, and can include the return of individual and/or aggregate research results to participants. There is strong support among funders, researchers, and participants for sharing aggregate results, which can include information about study populations and conclusions about groups of participants. Sharing aggregate results demonstrates respect and reciprocity, enhances public understanding of biomedical research, and may help to build trust in the research enterprise ? particularly in marginalized communities for whom historical and ongoing injustices have led to mistrust of clinicians and biomedical research. In genomics research specifically, communicating aggregate results may help to inform the public about the benefits and risks of broad data sharing, which is essential for advances in clinical genomics. Despite these merits, however, aggregate results sharing remains an uncommon practice in genomics research. Policies and best practices are urgently needed to ensure that aggregate results sharing becomes the norm rather than the exception, and to guide the development of materials that are appropriate and effective in diverse populations including those for low-literacy and non- English speaking participants. We propose a project that will develop and pilot test a collaborative, human-centered design approach to sharing aggregate research results in Spanish and English with socioeconomically diverse families participating in the Program in Prenatal and Pediatric Genomic Sequencing (P3EGS) at the University of California, San Francisco (UCSF). P3EGS is a member of the NIH-sponsored Clinical Sequencing Evidence-Generating Research (CSER) Consortium, whose six sites are studying the effectiveness of integrating genetic sequencing into the clinical care of ancestrally diverse and medically underserved patients.
P3EGS aims to assess the clinical use of exome sequencing for patients with a suspected genetic condition. Preliminary findings indicate that most P3EGS participants are interested in learning about aggregate results. In the proposed project, we aim to: 1) Identify aggregate results of interest to participants through direct engagement with 20 P3EGS participants and other stakeholder groups, including the CSER Stakeholder Advisory Council, 2) Develop electronic and paper materials using a collaborative approach involving repeated prototype development by a graphic artist with expertise in pairing stories and images, two rounds of interviews to gather feedback from 24 P3EGS participants, and consultation with the P3EGS research team and other key stakeholders, and 3) Assess the acceptability and relevance of the aggregate results materials through interviews with 12 P3EGS participants who received either the electronic or paper version. The long-term goal of the project is to inform future research and policies that support the development and implementation of aggregate results communication strategies that are effective across diverse populations.
Sharing aggregate research results, which can include overall study findings and conclusions about groups of participants, is an important way for researchers to show respect for research participants and convey new knowledge to the public. In clinical genomics research, however, sharing overall study results is still an uncommon practice, and not enough is known about how to share results in a manner that is acceptable, relevant, and understandable to participants. The proposed project will use a human-centered design strategy to develop and pilot test a method for communicating aggregate results to a diverse group of participants enrolled in a genetics research study.
