Background In the wake of mandates for biomedical research to increase participation by members of historically underrepresented populations, community engagement (CE) has emerged as a key intervention to help achieve this goal.Methods Using interviews, observations, and document analysis, we examine how stakeholders in precision medicine research understand and seek to put into practice ideas about who to engage, how engagement should be conducted, and what engagement is for.Results We find that ad hoc, opportunistic, and instrumental approaches to CE exacted significant (...) consequences for the time and resources devoted to engagement and the ultimate impacts it has on research. Critical differences emerged when engagement and research decisionmaking were integrated with each other versus occurring in parallel, separate parts of the study organization, and whether community members had the ability to determine which issues would be brought to them for consideration or to revise or even veto proposals made upstream based on criteria that mattered to them. CE was understood to have a range of purposes, from instrumentally facilitating recruitment and data collection, to advancing community priorities and concerns, to furthering long-term investments in relationships with and changes in communities. These choices about who to engage, what engagement activities to support, how to solicit and integrate community input into the workflow of the study, and what CE was for were often conditioned upon preexisting perceptions and upstream decisions about study goals, competing priorities, and resource availability.Conclusions Upstream choices about CE and constraints of time and resources cascade into tradeoffs that often culminated in “pantomime community engagement.” This approach can create downstream costs when engagement is experienced as improvised and sporadic. Transformations are needed for CE to be seen as a necessary scientific investment and part of the scientific process. (shrink)

Because no single person or group holds knowledge about all aspects of research, mechanisms are needed to support knowledge exchange and engagement. Expertise in the research setting necessarily includes scientific and methodological expertise, but also expertise gained through the experience of participating in research and/or being a recipient of research outcomes. Engagement is, by its nature, reciprocal and relational: the process of engaging research participants, patients, citizens and others brings them closer to the research but also brings the research closer (...) to them. When translating research into practice, engaging the public and other stakeholders is explicitly intended to make the outcomes of translation relevant to its constituency of users. In practice, engagement faces numerous challenges and is often time-consuming, expensive and ‘thorny’ work. We explore the epistemic and ontological considerations and implications of four common critiques of engagement methodologies that contest: representativeness, communication and articulation, impacts and outcome, and democracy. The ECOUTER methodology addresses problems of representation and epistemic foundationalism using a methodology that asks, “How could it be otherwise?” ECOUTER affords the possibility of engagement where spatial and temporal constraints are present, relying on saturation as a method of ‘keeping open’ the possible considerations that might emerge and including reflexive use of qualitative analytic methods. This paper describes the ECOUTER process, focusing on one worked example and detailing lessons learned from four other pilots. ECOUTER uses mind-mapping techniques to ‘open up’ engagement, iteratively and organically. ECOUTER aims to balance the breadth, accessibility and user-determination of the scope of engagement. An ECOUTER exercise comprises four stages: engagement and knowledge exchange; analysis of mindmap contributions; development of a conceptual schema ; and feedback, refinement and development of recommendations. ECOUTER refuses fixed truths but also refuses a fixed nature. Its promise lies in its flexibility, adaptability and openness. ECOUTER will be formed and re-formed by the needs and creativity of those who use it. (shrink)

During the past decades, research on ethical, legal, and social aspects of biobanks suggested and analysed various ethically and socially justifiable frameworks for collecting, storing, and distributing human biological material and bioinformation. In this article, we identify three patterns of argument that differ in terms of shared core assumptions and similar conceptual as well as normative orientations. These discursive 'orders', which are related to specific macropolitical contexts, have significantly shaped contexts for biobank policymaking. The first order was characterised by high (...) expectations of genomics and biobanking. Second order discourse partly took over the problems located in the first order, but reintroduced them into a justificatory framework that identified biobanks as public goods per se. The third order of ELSA expertise maintained a supportive attitude towards biobanking. However, regulation based on deductive reasoning became progressively complemented by ideals of participatory mechanisms and different methodologies of studying public perceptions. We conclude that this emphasis on learning processes and deliberation helps biobank communities to develop new concepts, methods and insights that will prove helpful in order to adapt to essentially undetermined futures of transnational innovation societies. (shrink)

Building a biobank network in developing countries is essential to foster genomic research and precision medicine for patients’ benefit. However, there are serious barriers to establishing biobanks in low-income and middle-income countries (LMICs), including Ukraine. Here, we outline key barriers and essential milestones for the successful expansion of biobanks, genomic research and personalised medicine in Ukraine, drawing from the experience of other LMICs. A lack of legal and ethical governance in conjunction with limited awareness about biobanking and community distrust are (...) the principal threats to establishing biobanks. The experiences of LMICs suggest that Ukraine urgently needs national guidelines covering ethical and legal aspects of biospecimen-related research. National guidelines must be consistent with international ethical recommendations for safeguarding participants’ rights, welfare and privacy. Additionally, efforts to educate and engage physicians and patient communities are essential for achieving biobanking goals and benefits for precision medicine and future patients. (shrink)

Public participation is increasingly an aspect of policy development in many areas, and the governance of biomedical research is no exception. There are good reasons for this: biomedical research relies on public funding; it relies on biological samples and information from large numbers of patients and healthy individuals; and the outcomes of biomedical research are dramatically and irrevocably changing our society. There is thus arguably a democratic imperative for including public values in strategic decisions about the governance of biomedical research. (...) However, it is not immediately clear how this might best be achieved. While different approaches have been proposed and trialled, we focus here on the use of public deliberation as a mechanism to develop input for policy on biomedical research. We begin by explaining the rationale for conducting public deliberation in biomedical research. We focus, in particular, on the ELS (ethical, legal, social) aspects of human tissue biobanking. The last few years have seen the development of methods for conducting public deliberation on these issues in several jurisdictions, for the purpose of incorporating lay public voices in biobanking policy. We explain the theoretical foundation underlying the notion of deliberation, and outline the main lessons and capacities that have been developed in the area of conducting public deliberation on biobanks. We next provide an analysis of the theoretical and practical challenges that we feel still need to be addressed for the use of public deliberation to guide ethical norms and governance of biomedical research. We examine the issues of: (i) linking the outcomes of deliberation to tangible action; (ii) the mandate under which a deliberation is conducted; (iii) the relative weight that should be accorded to a public deliberative forum vs other relevant voices; (iv) evaluating the quality of deliberation; and (5) the problem of scalability of minipublics. (shrink)

BackgroundEngagement is important within cohort studies for a number of reasons. It is argued that engaging participants within the studies they are involved in may promote their recruitment and retention within the studies. Participant input can also improve study designs, make them more acceptable for uptake by participants and aid in contextualising research communication to participants. Ultimately it is also argued that engagement needs to provide an avenue for participants to feedback to the cohort study and that this is an (...) ethical imperative. This study sought to explore the participants’ experiences and thoughts of their engagement with their birth cohort study.MethodsParticipants were recruited from the Children of the 90s (CO90s) study. Qualitative semi-structured interviews were conducted with 42 participants. The interviews were transcribed verbatim, and uploaded onto Nvivo software. They were then analysed via thematic analysis with a constant comparison technique.ResultsParticipants’ experiences of their engagement with CO90s were broadly based on three aspects: communication they received from CO90s, experiences of ethical conduct from CO90s and receiving rewards from CO90s. The communication received from CO90s, ranged from newsletters explaining study findings and future studies, to more personal forms like annual greeting cards posted to each participant. Ethical conduct from CO90s mainly involved participants understanding that CO90s would keep their information confidential, that it was only involved in ‘good’ ethical research and their expectation that CO90s would always prioritise participant welfare. Some of the gifts participants said they received at CO90s included toys, shopping vouchers, results from clinical tests, and time off from school to attend data collection (Focus) days. Participants also described a temporality in their engagement with CO90s and the subsequent trust they had developed for the cohort study.ConclusionThe experiences of engagement described by participants were theorized as being based on reciprocity which was sometimes overt and other times more nuanced. We further provide empirical evidence of participants’ expectation for a reciprocal interaction with their cohort study while highlighting the trust that such an interaction fosters. Our study therefore provides key insights for other cohort studies on what participants value in their interactions with their cohort studies. (shrink)

Biobanks are precariously situated at the intersection of science, genetics, genomics, society, ethics, the law and politics. This multi-disciplinarity has given rise to a new discourse in health research involving diverse stakeholders. Each stakeholder is embedded in a unique context and articulates his/her biobanking activities differently. To researchers, biobanks carry enormous transformative potential in terms of advancing scientific discovery and knowledge. However, in the context of power asymmetries in Africa and a distrust in science born out of historical exploitation, researchers (...) must balance the scientific imperative of collecting, storing and sharing high quality biological samples with obligations to donors/participants, communities, international collaborators, regulatory and ethics authorities. To date, researcher perspectives on biobanking in South Africa have not been explored and documented. In-depth qualitative interviews were conducted with a purposive sample of 21 researchers – 8 in the Western Cape, 3 in Gauteng and 10 in Kwa-Zulu Natal. Interviews lasted approximately 40–60 min and were audiotaped with consent. Thematic analysis of the transcribed interviews was conducted by the co-authors. Researchers articulated serious concerns over standardised regulatory approaches that failed to consider the heterogeneity of biobanks. Given that biobanks differ considerably, guidelines and RECs need to stratify risk accordingly and governance processes and structures must be flexible. While RECs were regarded as an important component of the governance structure researchers expressed concern about their expertise in biobanking. Operational management of biobanks was regarded as an ethical imperative and a pre-requisite to building trust during consent processes. While broad general consent was preferred, tiered consent was thought to be more consistent with respect for autonomy and building trust. Material Transfer Agreements were often lacking when biosamples were exported and this was perceived to impact negatively on trust. On the other hand, researchers believed that authentic community engagement would help to build trust. Building trust will best be achieved via a system of governance structures and processes that precede the establishment of a biobank and monitor progress from the point of sample collection through to future use, including export. Such governance structures must be robust and must include comprehensive national legislation, policy and contextualised guidelines. Currently such governance infrastructure appears to be lacking in many African countries including South Africa. Capacity development of all stakeholders including REC members will enhance expeditious and efficient review of biobanking protocols which in turn will reinforce trust in the researcher-donor relationship. Science translation and community engagement in biobanking is integral to the success of biobanking in South Africa. (shrink)

Genetic research into ageing, longevity and late-onset disease is becoming increasingly common. Yet, there is a paucity of knowledge related to clinical actionability and the return of pathogenic variants to otherwise healthy elderly individuals. Whether or not genetic research in the elderly should be managed differently from standard practices adapted for younger populations has not yet been defined. In this article, we provide an overview of ethical and practical challenges in preparing for a genetic study of over 14 000 healthy (...) Australians aged 70 years or older enrolled in the ASPirin in Reducing Events in the Elderly Healthy Ageing Biobank. At the time of consent, all participants in this study were free of life-threatening illness, cardiovascular disease or cognitive impairment. ASPREE is thus a cohort of healthy elderly individuals with seemingly minimal burden of genetic disease recruited without ascertainment bias. The cohort presents a unique opportunity to address the penetrance of known pathogenic variants in a population without disease symptoms; however, it also raises a number of ethical concerns regarding the interpretation and disclosure of variants with known clinical actionability. Some of the challenges include how to manage the interpretation, disclosure and actioning of pathogenic variants found in otherwise healthy elderly adults without disease symptoms, whether or not to disclose findings for the benefit of family members rather than elderly consented donors themselves, how to manage the return of genetic findings to the elderly individuals who are now in severe cognitive decline or terminal illness, how to ensure quality of information and clinical service upon disclosure of results to this demographic and how to prepare for the insurance implications of disclosing genetic information under Australian law. We discuss these and other dilemmas and propose a defensible plan of management. Trial registration number ISRCTN83772183. (shrink)

Different types of consent are used to obtain human biospecimens for future research. This variation has resulted in confusion regarding what research is permitted, inadvertent constraints on future research, and research proceeding without consent. The National Institutes of Health Clinical Center's Department of Bioethics held a workshop to consider the ethical acceptability of addressing these concerns by using broad consent for future research on stored biospecimens. Multiple bioethics scholars, who have written on these issues, discussed the reasons for consent, the (...) range of consent strategies, and gaps in our understanding, and concluded with a proposal for broad initial consent coupled with oversight and, when feasible, ongoing provision of information to donors. This article describes areas of agreement and areas that need more research and dialogue. Given recent proposed changes to the Common Rule, and new guidance regarding storing and sharing data and samples, this is an important and tim.. (shrink)