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Return of individual research results from genomic research: A systematic review of stakeholder perspectives

ABSTRACT: Despite the plethora of empirical studies conducted to date, debate continues about whether and to what extent results should be returned to participants of genomic research. We aimed to systematically review the empirical literature exploring stakeholders’ perspectives on return of individual research results (IRR) from genomic research. We examined preferences for receiving or willingness to return IRR, and experiences with either receiving or returning them. The systematic searches were conducted across five major databases in August 2018 and repeated in April 2020, and included studies reporting findings from primary research regardless of method (quantitative, qualitative, mixed). Articles that related to the clinical setting were excluded. Our search identified 221 articles that met our search criteria. This included 118 quantitative, 69 qualitative and 34 mixed methods studies. These articles included a total number of 118,874 stakeholders with research participants (85,270/72%) and members of the general public (40,967/35%) being the largest groups represented. The articles spanned at least 22 different countries with most (144/65%) being from the USA. Most (76%) discussed clinical research projects, rather than biobanks. More than half (58%) gauged views that were hypothetical. We found overwhelming evidence of high interest in return of IRR from potential and actual genomic research participants. There is also a general willingness to provide such results by researchers and health professionals, although they tend to adopt a more cautious stance. While all results are desired to some degree, those that have the potential to change clinical management are generally prioritized by all stakeholders. Professional stakeholders appear more willing to return results that are reliable and clinically relevant than those that are less reliable and lack clinical relevance. The lack of evidence for significant enduring psychological harm and the clear benefits to some research participants suggest that researchers should be returning actionable IRRs to participants.

SUBMITTER: Vears D

PROVIDER: S-EPMC8575249 | biostudies-literature |

REPOSITORIES: biostudies-literature

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Project description:Newer data platforms offer increased opportunity to share multidimensional health data with research participants, but the preferences of participants for which data to receive and how is evolving. Our objective is to describe the preferences and expectations of participants for the return of individual research results within Project Baseline Health Study (PBHS). The PBHS is an ongoing, multicenter, longitudinal cohort study with data from four initial enrollment sites. PBHS participants are recruited from the general population along with groups enriched for heart disease and cancer disease risk. Cross-sectional data on return of results were collected in 2017-2018 from an (1) in-person enrollment survey (n = 1,890), (2) benchmark online survey (n = 1,059), and (3) participant interviews (n = 21). The main outcomes included (1) preferences for type of information to be added next to returned results, (2) participant plans for sharing returned results with a non-study clinician, and (3) choice to opt-out of receiving genetic results. Results were compared by sociodemographic characteristics. Enrollment and benchmark survey respondents were 57.1% and 53.5% female, and 60.0% and 66.2% white, respectively. Participants preferred the following data types be added to returned results in the future: genetics (29.9%), heart imaging, (16.4%), study watch (15.8%), and microbiome (13.3%). Older adults (OR 0.60, 95% CI: 0.41-0.87) were less likely to want their genetic results returned next. Forty percent of participants reported that they would not share all returned results with their non-study clinicians. Black (OR 0.64, 95% CI 0.43-0.95) and Asian (OR 0.47, 95% CI 0.30-0.73) participants were less likely, and older participants more likely (OR 1.45-1.61), to plan to share all results with their clinician than their counterparts. At enrollment, 5.8% of participants opted out of receiving their genetics results. The study showed that substantial heterogeneity existed in participant's preferences and expectations for return of results, and variations were related to sociodemographic characteristics.

Project description:ObjectivesThe purpose of this study was to partner with stakeholders to identify gaps in care for persons living with dementia and their family caregivers and from this list, identify priorities for dementia care research.MethodsUsing a community-engaged research approach, a Stakeholder Advisory Council (SAC) consisting of diverse membership including persons living with dementia and family caregivers was convened. Through our work with the SAC, along with input from the wider network through a symposium, webinars, and an online learning community, gaps in dementia care and a list of topics for dementia care research was generated. This list was reduced to 46 topics for dementia care research and sent to stakeholders (persons living with dementia, family caregivers, and health/social care professionals in dementia care) to be prioritized by rating each of the 46 topics as "Not so important," "Important," or "Very important." Priorities for dementia care were summarized by frequencies and proportions.ResultsA total of 186 participants completed the survey from August through October 2020, including 23 (12.4%) persons living with dementia, 101 (54.3%) family caregivers, and 62 (33.3%) health/social care professionals. Consistent across stakeholder groups was the focus on research on how best to support families following a diagnosis of dementia. Among persons living with dementia, research focused on support for continuing to live in their own homes was ranked as the highest priority, rated by 91.3% as "Very Important". High priority research areas for family caregivers included interventions to slow cognitive decline (76.3%) as well as non-pharmacological approaches to manage behavioral symptoms (74.7%). The highest priority research topics for health/social care professionals were focused on the diagnosis including benefits of an early diagnosis (71.4%), how best to deliver the diagnosis (70.9%), and supports needed following a diagnosis (78.6%).ConclusionsThis project draws on the strengths of its multi-stakeholder perspective to support patient-centered outcomes research. Findings are intended to inform those who conduct research and those who fund research about which research topics stakeholders believe are most important and thus have greatest potential to improve the quality of life among people living with dementia and their families.

Project description:Participatory research approaches have developed in response to the growing emphasis on translation of research evidence into practice. However, there are few published examples of stakeholder engagement strategies, and little guidance specific to larger ongoing research programs or those with a rural focus. This paper describes the evolution, structure, and processes of an annual Rural Dementia Summit launched in 2008 as an engagement strategy for the Rural Dementia Action Research (RaDAR) program and ongoing for more than 10 years; and reports findings from a parallel mixed-methods study that includes stakeholder and researcher perspectives on the Summit's value and impact. Twelve years of stakeholder evaluations were analyzed. Rating scale data were summarized with descriptive statistics; open-ended questions were analyzed using an inductive thematic analysis. A thematic analysis was also used to analyze interviews with RaDAR researchers. Rating scale data showed high stakeholder satisfaction with all aspects of the Summit. Five themes were identified in the qualitative data: hearing diverse perspectives, building connections, collaborating for change, developing research and practice capacity, and leaving recharged. Five themes were identified in the researcher data: impact on development as a researcher, understanding stakeholder needs, informing research design, deepening commitment to rural dementia research, and building a culture of engagement. These findings reflect the key principles and impacts of stakeholder engagement reported in the literature. Additional findings include the value stakeholders place on connecting with stakeholders from diverse backgrounds, how the Summit was revitalizing, and how it developed stakeholder capacity to support change in their communities. Findings indicate that the Summit has developed into a community of practice where people with a common interest come together to learn and collaborate to improve rural dementia care. The Summit's success and sustainability are linked to RaDAR's responsiveness to stakeholder needs, the trust that has been established, and the value that stakeholders and researchers find in their participation.

Project description:BackgroundAdaptive clinical trials (ACTs) represent an emerging approach to trial design where accumulating data are used to make decisions about future conduct. Adaptations can include comparisons of multiple dose tiers, response-adaptive randomization, sample size re-estimation, and efficacy/futility stopping rules. The objective of this scoping review is to assess stakeholder attitudes, perspectives, and understanding of adaptive trials.MethodsWe conducted a review of articles examining stakeholders encompassing the broad medical trial community's perspectives of adaptive designs (ADs). A computerized search was conducted of four electronic databases with relevant search terms. Following screening of articles, the primary findings of each included article were coded for study design, population studied, purpose, and primary implications.ResultsOur team retrieved 167 peer-reviewed titles in total from the database search and 5 additional titles through searching web-based search engines for gray literature. Of those 172 titles, 152 were non-duplicate citations. Of these, 119 were not given full-text reviews, as their titles and abstracts indicated that they did not meet the inclusion criteria. Thirty-three articles were carefully examined for relevance, and of those, 18 were chosen to be part of the analysis; the other 15 were excluded, as they were not relevant upon closer inspection. Perceived advantages to ADs included limiting ineffective treatments and efficiency in answering the research question; -perceived barriers included insufficient sample size for secondary outcomes, challenges of consent, potential for bias, risk of type 1 error, cost and time to adaptively design trials, unclear rationales for using Ads, and, most importantly, a lack of education regarding ADs among stakeholders within the clinical trial community. Perceptions among different types of stakeholders varied from sector to sector, with patient perspectives being noticeably absent from the literature.ConclusionThere are diverse perceptions regarding ADs among stakeholders. Further training, guidelines, and toolkits on the proper use of ADs are needed at all levels to overcome many of these perceived barriers. While education for principal investigators is important, it is also crucial to educate other groups in the community, such as patients, as well as clinicians and staff involved in their daily implementation.

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Return of individual research results from genomic research: A systematic review of stakeholder perspectives

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