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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., Competing Interests: The authors have declared that no competing interests exist.

Background: Genomic and biosocial research data about individuals is rapidly proliferating, bringing the potential for novel opportunities for data integration and use. The scale, pace and novelty of these applications raise a number of urgent sociotechnical, ethical and legal questions, including optimal methods of data storage, management and access. Although the open science movement advocates unfettered access to research data, many of the UK's longitudinal cohort studies operate systems of managed data access, in which access is governed by legal and ethical agreements between stewards of research datasets and researchers wishing to make use of them. Amongst other things, these agreements aim to respect the reasonable expectations of the research participants who provided data and samples, as expressed in the consent process. Arguably, responsible data management and governance of data and sample use are foundational to the consent process in longitudinal studies and are an important source of trustworthiness in the eyes of those who contribute data to genomic and biosocial research., Methods: This paper presents an ethnographic case study exploring the foundational principles of a governance infrastructure for Managing Ethico-social, Technical and Administrative issues in Data ACcess (METADAC), which are operationalised through a committee known as the METADAC Access Committee. METADAC governs access to phenotype, genotype and 'omic' data and samples from five UK longitudinal studies., Findings: Using the example of METADAC, we argue that three key structural features are foundational for practising responsible data sharing: independence and transparency; interdisciplinarity; and participant-centric decision-making. We observe that the international research community is proactively working towards optimising the use of research data, integrating/linking these data with routine data generated by health and social care services and other administrative data services to improve the analysis, interpretation and utility of these data. The governance of these new complex data assemblages will require a range of expertise from across a number of domains and disciplines, including that of study participants. Human-mediated decision-making bodies will be central to ensuring achievable, reasoned and responsible decisions about the use of these data; the METADAC model described in this paper provides an example of how this could be realised.

Background: Governments, funding bodies, institutions, and publishers have developed a number of strategies to encourage researchers to facilitate access to datasets. The rationale behind this approach is that this will bring a number of benefits and enable advances in healthcare and medicine by allowing the maximum returns from the investment in research, as well as reducing waste and promoting transparency. As this approach gains momentum, these data-sharing practices have implications for many kinds of research as they become standard practice across the world., Main Text: The governance frameworks that have been developed to support biomedical research are not well equipped to deal with the complexities of international data sharing. This system is nationally based and is dependent upon expert committees for oversight and compliance, which has often led to piece-meal decision-making. This system tends to perpetuate inequalities by obscuring the contributions and the important role of different data providers along the data stream, whether they be low- or middle-income country researchers, patients, research participants, groups, or communities. As research and data-sharing activities are largely publicly funded, there is a strong moral argument for including the people who provide the data in decision-making and to develop governance systems for their continued participation., Conclusions: We recommend that governance of science becomes more transparent, representative, and responsive to the voices of many constituencies by conducting public consultations about data-sharing addressing issues of access and use; including all data providers in decision-making about the use and sharing of data along the whole of the data stream; and using digital technologies to encourage accessibility, transparency, and accountability. We anticipate that this approach could enhance the legitimacy of the research process, generate insights that may otherwise be overlooked or ignored, and help to bring valuable perspectives into the decision-making around international data sharing.

The social structures that govern data/sample release aim to safeguard the confidentiality and privacy of cohort research participants (without whom there would be no data or samples) and enable the realization of societal benefit through optimizing the scientific use of those cohorts. Within collaborations involving multiple cohorts and biobanks, however, the local, national, and supranational institutional and legal guidelines for research (which produce a multiplicity of data access governance structures and guidelines) risk impeding the very science that is the raison d'etre of these consortia. We present an ethnographic study, which examined the epistemic and nonepistemic values driving decisions about data access and their consequences in the context of the pilot of an integrated approach to co-analysis of data. We demonstrate how the potential analytic flexibility offered by this approach was lost under contemporary data access governance. We identify three dominant values: protecting the research participant, protecting the study, and protecting the researcher. These values were both supported by and juxtaposed against a "public good" argument, and each was used as a rationale to both promote and inhibit sharing of data. While protection of the research participants was central to access permissions, decisions were also attentive to the desire of researchers to see their efforts in building population biobanks and cohorts realized in the form of scientific outputs. We conclude that systems for governing and enabling data access in large consortia need to (1) protect disclosure of research participant information or identity, (2) ensure the specific expectations of research participants are met, (3) embody systems of review that are transparent and not compromised by the specific interests of one particular group of stakeholders, and (4) facilitate data access procedures that are timely and efficient. Practical solutions are urgently needed. New approaches to data access governance should be trialed (and formally evaluated) with input from and discussion with stakeholders.

Motivation: The data that put the 'evidence' into 'evidence-based medicine' are central to developments in public health, primary and hospital care. A fundamental challenge is to site such data in repositories that can easily be accessed under appropriate technical and governance controls which are effectively audited and are viewed as trustworthy by diverse stakeholders. This demands socio-technical solutions that may easily become enmeshed in protracted debate and controversy as they encounter the norms, values, expectations and concerns of diverse stakeholders. In this context, the development of what are called 'Data Safe Havens' has been crucial. Unfortunately, the origins and evolution of the term have led to a range of different definitions being assumed by different groups. There is, however, an intuitively meaningful interpretation that is often assumed by those who have not previously encountered the term: a repository in which useful but potentially sensitive data may be kept securely under governance and informatics systems that are fit-for-purpose and appropriately tailored to the nature of the data being maintained, and may be accessed and utilized by legitimate users undertaking work and research contributing to biomedicine, health and/or to ongoing development of healthcare systems., Results: This review explores a fundamental question: 'what are the specific criteria that ought reasonably to be met by a data repository if it is to be seen as consistent with this interpretation and viewed as worthy of being accorded the status of 'Data Safe Haven' by key stakeholders'? We propose 12 such criteria., Contact: paul.burton@bristol.ac.uk., (© The Author 2015. Published by Oxford University Press.)

Background: DataSHIELD (Data Aggregation Through Anonymous Summary-statistics from Harmonised Individual levEL Databases) has been proposed to facilitate the co-analysis of individual-level data from multiple studies without physically sharing the data. In a previous paper, we investigated whether DataSHIELD could protect participant confidentiality in accordance with UK law. In this follow-up paper, we investigate whether DataSHIELD addresses a broader range of ethics-related data-sharing concerns., Methods: Ethics-related data-sharing concerns of Institutional Review Boards, ethics experts, international research consortia and research participants were identified through a literature search and systematically examined at a multidisciplinary workshop to determine whether DataSHIELD proposes mechanisms which can address these concerns., Results: DataSHIELD addresses several ethics-related data-sharing concerns related to privacy, confidentiality, and the protection of the research participant's rights while sharing data and after the data have been shared. The data remain entirely under the direct management of the study that collected them. Data processing commands are strictly supervised, and the data are queried in a protected environment. Issues related to the return of individual research results when data are shared are eliminated; the responsibility for return remains at the study of origin., Conclusion: DataSHIELD can provide an innovative and robust solution for addressing commonly encountered ethics-related data-sharing concerns., (© 2014 S. Karger AG, Basel.)

Background: Research in modern biomedicine and social science requires sample sizes so large that they can often only be achieved through a pooled co-analysis of data from several studies. But the pooling of information from individuals in a central database that may be queried by researchers raises important ethico-legal questions and can be controversial. In the UK this has been highlighted by recent debate and controversy relating to the UK's proposed 'care.data' initiative, and these issues reflect important societal and professional concerns about privacy, confidentiality and intellectual property. DataSHIELD provides a novel technological solution that can circumvent some of the most basic challenges in facilitating the access of researchers and other healthcare professionals to individual-level data., Methods: Commands are sent from a central analysis computer (AC) to several data computers (DCs) storing the data to be co-analysed. The data sets are analysed simultaneously but in parallel. The separate parallelized analyses are linked by non-disclosive summary statistics and commands transmitted back and forth between the DCs and the AC. This paper describes the technical implementation of DataSHIELD using a modified R statistical environment linked to an Opal database deployed behind the computer firewall of each DC. Analysis is controlled through a standard R environment at the AC., Results: Based on this Opal/R implementation, DataSHIELD is currently used by the Healthy Obese Project and the Environmental Core Project (BioSHaRE-EU) for the federated analysis of 10 data sets across eight European countries, and this illustrates the opportunities and challenges presented by the DataSHIELD approach., Conclusions: DataSHIELD facilitates important research in settings where: (i) a co-analysis of individual-level data from several studies is scientifically necessary but governance restrictions prohibit the release or sharing of some of the required data, and/or render data access unacceptably slow; (ii) a research group (e.g. in a developing nation) is particularly vulnerable to loss of intellectual property-the researchers want to fully share the information held in their data with national and international collaborators, but do not wish to hand over the physical data themselves; and (iii) a data set is to be included in an individual-level co-analysis but the physical size of the data precludes direct transfer to a new site for analysis., (© The Author 2014; all rights reserved. Published by Oxford University Press on behalf of the International Epidemiological Association.)

Study Objective: In the context of calls to develop better systems for out-of-hospital clinical research, we seek to understand paramedics' perceptions of involvement in research and the barriers and facilitators to that involvement., Methods: This was a qualitative study using semistructured focus groups with 58 United Kingdom paramedics and interviews with 30 US firefighter-paramedics. The study focused on out-of-hospital research (trials of out-of-hospital treatment for stroke), whereby paramedics identified potential study subjects or obtained consent and administered study treatment in the field. Data were analyzed with a thematic and discourse approach., Results: Three key themes emerged as significant facilitators and barriers to paramedic involvement in research: patient benefit, professional identity and responsibility, and time. Paramedics showed willingness and capacity to engage in research but also some reticence because of the perceived sacrifice of autonomy and challenge to their identity. Paramedics work in a time-sensitive environment and were concerned that research would increase time taken in the field., Conclusion: Awareness of these perspectives will help with development of out-of-hospital research protocols and potentially facilitate greater participation., (Copyright © 2012 American College of Emergency Physicians. Published by Mosby, Inc. All rights reserved.)