What does engagement mean to participants in longitudinal cohort studies? A qualitative study.

BACKGROUND: Engagement 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. METHODS: Participants 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. RESULTS: Participants' 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. CONCLUSION: The 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.

APPLaUD: access for patients and participants to individual level uninterpreted genomic data.

BACKGROUND: There is a growing support for the stance that patients and research participants should have better and easier access to their raw (uninterpreted) genomic sequence data in both clinical and research contexts. MAIN BODY: We review legal frameworks and literature on the benefits, risks, and practical barriers of providing individuals access to their data. We also survey genomic sequencing initiatives that provide or plan to provide individual access. Many patients and research participants expect to be able to access their health and genomic data. Individuals have a legal right to access their genomic data in some countries and contexts. Moreover, increasing numbers of participatory research projects, direct-to-consumer genetic testing companies, and now major national sequencing initiatives grant individuals access to their genomic sequence data upon request. CONCLUSION: Drawing on current practice and regulatory analysis, we outline legal, ethical, and practical guidance for genomic sequencing initiatives seeking to offer interested patients and participants access to their raw genomic data.

Including all voices in international data-sharing governance.

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.

Better governance, better access: practising responsible data sharing in the METADAC governance infrastructure.

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.

Smart homes, private homes? An empirical study of technology researchers' perceptions of ethical issues in developing smart-home health technologies.

BACKGROUND: Smart-home technologies, comprising environmental sensors, wearables and video are attracting interest in home healthcare delivery. Development of such technology is usually justified on the basis of the technology's potential to increase the autonomy of people living with long-term conditions. Studies of the ethics of smart-homes raise concerns about privacy, consent, social isolation and equity of access. Few studies have investigated the ethical perspectives of smart-home engineers themselves. By exploring the views of engineering researchers in a large smart-home project, we sought to contribute to dialogue between ethics and the engineering community. METHODS: Either face-to-face or using Skype, we conducted in-depth qualitative interviews with 20 early- and mid-career smart-home researchers from a multi-centre smart-home project, who were asked to describe their own experience and to reflect more broadly about ethical considerations that relate to smart-home design. With participants' consent, interviews were audio-recorded, transcribed and analysed using a thematic approach. RESULTS: Two overarching themes emerged: in 'Privacy', researchers indicated that they paid close attention to negative consequences of potential unauthorised information sharing in their current work. However, when discussing broader issues in smart-home design beyond the confines of their immediate project, researchers considered physical privacy to a lesser extent, even though physical privacy may manifest in emotive concerns about being watched or monitored. In 'Choice', researchers indicated they often saw provision of choice to end-users as a solution to ethical dilemmas. While researchers indicated that choices of end-users may need to be restricted for technological reasons, ethical standpoints that restrict choice were usually assumed and embedded in design. CONCLUSIONS: The tractability of informational privacy may explain the greater attention that is paid to it. However, concerns about physical privacy may reduce acceptability of smart-home technologies to future end-users. While attention to choice suggests links with privacy, this may misidentify the sources of privacy and risk unjustly burdening end-users with problems that they cannot resolve. Separating considerations of choice and privacy may result in more satisfactory treatment of both. Finally, through our engagement with researchers as participants this study demonstrates the relevance of (bio)ethics as a critical partner to smart-home engineering.

The ECOUTER methodology for stakeholder engagement in translational research.

BACKGROUND: 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 (as a patient or member of the public). Engagement is, by its nature, reciprocal and relational: the process of engaging research participants, patients, citizens and others (the many 'publics' of engagement) 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. METHODS: 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 (Employing COnceptUal schema for policy and Translation Engagement in Research) 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. RESULTS: 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: (1) engagement and knowledge exchange; (2) analysis of mindmap contributions; (3) development of a conceptual schema (i.e. a map of concepts and their relationship); and (4) feedback, refinement and development of recommendations. CONCLUSION: 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.

Data Safe Havens in health research and healthcare.

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.

Treating childhood intermittent distance exotropia: a qualitative study of decision making.

BACKGROUND: Engaging patients (parents/families) in treatment decisions is increasingly recognised as important and beneficial. Yet where the evidence base for treatment options is limited, as with intermittent distance exotropia (X(T)), this presents a challenge for families and clinicians. The purpose of this study was to explore how decisions are made in the management and treatment of X(T) and what can be done to support decision-making for clinicians, parents and children. METHODS: This was a qualitative study using face to face interviews with consultant ophthalmologists and orthoptists, and parents of children with X(T). Interview data were analysed using the constant comparative method. RESULTS: The drivers for clinicians in treatment decision-making for X(T) were the proportion of time the strabismus is manifest and parents' views. For parents, decisions were influenced by: fear of bullying and, to a lesser degree, concerns around the impact of the strabismus on their child's vision. Uncertainty around the effectiveness of treatment options caused difficulties for some clinicians when communicating with parents. Parental understanding of the nature of X(T) and rationale for treatment often differed from that of the clinicians, and this affected their involvement in decision-making. Though there were good examples of shared decision-making and parent and child engagement some parents said the process felt rushed and they felt excluded. Parents reported that clinicians provided sufficient information in consultations but they had difficulties in retaining verbal information to convey to other family members. CONCLUSIONS: Overall parents were happy with the care their child received but there is scope for better parent and (where appropriate) child engagement in decision-making. There was an expressed need for written information about X(T) to reinforce what was given verbally in consultations and to share with other family members. Access could be via the hospital website, along with videos or blogs from parents and children who have undergone the various management options. A method of assisting clinicians to explain the treatment options, together with the uncertainties, in a clear and concise way could be of particular benefit to orthoptists who have the most regular contact with parents and children, and are more likely to suggest conservative treatments such as occlusion and minus lenses.

The prevalence of metabolic syndrome and metabolically healthy obesity in Europe: a collaborative analysis of ten large cohort studies.

BACKGROUND: Not all obese subjects have an adverse metabolic profile predisposing them to developing type 2 diabetes or cardiovascular disease. The BioSHaRE-EU Healthy Obese Project aims to gain insights into the consequences of (healthy) obesity using data on risk factors and phenotypes across several large-scale cohort studies. Aim of this study was to describe the prevalence of obesity, metabolic syndrome (MetS) and metabolically healthy obesity (MHO) in ten participating studies. METHODS: Ten different cohorts in seven countries were combined, using data transformed into a harmonized format. All participants were of European origin, with age 18-80 years. They had participated in a clinical examination for anthropometric and blood pressure measurements. Blood samples had been drawn for analysis of lipids and glucose. Presence of MetS was assessed in those with obesity (BMI ≥ 30 kg/m2) based on the 2001 NCEP ATP III criteria, as well as an adapted set of less strict criteria. MHO was defined as obesity, having none of the MetS components, and no previous diagnosis of cardiovascular disease. RESULTS: Data for 163,517 individuals were available; 17% were obese (11,465 men and 16,612 women). The prevalence of obesity varied from 11.6% in the Italian CHRIS cohort to 26.3% in the German KORA cohort. The age-standardized percentage of obese subjects with MetS ranged in women from 24% in CHRIS to 65% in the Finnish Health2000 cohort, and in men from 43% in CHRIS to 78% in the Finnish DILGOM cohort, with elevated blood pressure the most frequently occurring factor contributing to the prevalence of the metabolic syndrome. The age-standardized prevalence of MHO varied in women from 7% in Health2000 to 28% in NCDS, and in men from 2% in DILGOM to 19% in CHRIS. MHO was more prevalent in women than in men, and decreased with age in both sexes. CONCLUSIONS: Through a rigorous harmonization process, the BioSHaRE-EU consortium was able to compare key characteristics defining the metabolically healthy obese phenotype across ten cohort studies. There is considerable variability in the prevalence of healthy obesity across the different European populations studied, even when unified criteria were used to classify this phenotype.

Health state descriptions to elicit stroke values: do they reflect patient experience of stroke?

BACKGROUND: To explore whether stroke health state descriptions used in preference elicitation studies reflect patients' experiences by comparing published descriptions with qualitative studies exploring patients' lived experience. METHODS: Two literature reviews were conducted: on stroke health state descriptions used in direct preference elicitation studies and the qualitative literature on patients' stroke experience. Content and comparative thematic analysis was used to identify characteristics of stroke experience in both types of study which were further mapped onto health related quality of life (HRQOL) domains relevant to stroke. Two authors reviewed the coded text, categories and domains. RESULTS: We included 35 studies: seven direct preference elicitation studies and 28 qualitative studies on patients' experience. Fifteen coded categories were identified in the published health state descriptions and 29 in the qualitative studies. When mapped onto domains related to HRQOL, qualitative studies included a wider range of categories in every domain that were relevant to the patients' experience than health state descriptions. CONCLUSIONS: Variation exists in the content of health state descriptions for all levels of stroke severity, most critically with a major disjuncture between the content of descriptions and how stroke is experienced by patients. There is no systematic method for constructing the content/scope of health state descriptions for stroke, and the patient perspective is not incorporated, producing descriptions with major deficits in reflecting the lived experience of stroke, and raising serious questions about the values derived from such descriptions and conclusions based on these values.

Reconciling the biomedical data commons and the GDPR: three lessons from the EUCAN ELSI collaboratory.

The coming-into-force of the EU General Data Protection Regulation (GDPR) is a watershed moment in the legal recognition of enforceable rights to informational self-determination. The rapid evolution of legal requirements applicable to data use, however, has the potential to outstrip the capabilities of networks of biomedical data users to respond to the shifting norms. It can also delegitimate established institutional bodies that are responsible for assessing and authorising the downstream use of data, including research ethics committees and institutional data custodians. These burdens are especially pronounced for clinical and research networks that are of transnational scale, because the legal compliance burden for outbound international data transfers from the EEA is especially high. Legislatures, courts, and regulators in the EU should therefore implement the following three legal changes. First, the responsibilities of particular actors in a data sharing network should be delimited through the contractual allocation of responsibilities between collaborators. Second, the use of data through secure data processing environments should not trigger the international transfer provisions of the GDPR. Third, the use of federated data analysis methodologies that do not provide analysis nodes or downstream users access to identifiable personal data as part of the outputs of those analyses should not be considered circumstances of joint controllership, nor lead to the users of non-identifiable data to be considered controllers or processors. These small clarifications of, or modifications to, the GDPR would facilitate the exchange of biomedical data amongst clinicians and researchers.

The impact of the UK 'Act FAST' stroke awareness campaign: content analysis of patients, witness and primary care clinicians' perceptions.

BACKGROUND: The English mass media campaign 'Act FAST' aimed to raise stroke awareness and the need to call emergency services at the onset of suspected stroke. We examined the perceived impact and views of the campaign in target populations to identify potential ways to optimise mass-media interventions for stroke. METHODS: Analysis of semi-structured interviews conducted as part of two qualitative studies, which examined factors influencing patient/witness response to acute stroke symptoms (n = 19 stroke patients, n = 26 stroke witnesses) and perceptions about raising stroke awareness in primary care (n = 30 clinicians). Both studies included questions about the 'Act FAST' campaign. Interviews were content analysed to determine campaign awareness, perceived impact on decisions and response to stroke, and views of the campaign. RESULTS: Most participants were aware of the Act FAST campaign. Some patients and witnesses reported that the campaign impacted upon their stroke recognition and response, but the majority reported no impact. Clinicians often perceived campaign success in raising stroke awareness, but few thought it would change response behaviours. Some patients and witnesses, and most primary care clinicians expressed positive views towards the campaign. Some more critical participant comments included perceptions of dramatic, irrelevant, and potentially confusing content, such as a prominent 'fire in the brain' analogy. CONCLUSIONS: Act FAST has had some perceived impact on stroke recognition and response in some stroke patients and witnesses, but the majority reported no campaign impact. Primary care clinicians were positive about the campaign, and believed it had impacted on stroke awareness and recognition but doubted impact on response behaviour. Potential avenues for optimising and complementing mass media campaigns such as 'Act FAST' were identified.

A review of decision support, risk communication and patient information tools for thrombolytic treatment in acute stroke: lessons for tool developers.

BACKGROUND: Tools to support clinical or patient decision-making in the treatment/management of a health condition are used in a range of clinical settings for numerous preference-sensitive healthcare decisions. Their impact in clinical practice is largely dependent on their quality across a range of domains. We critically analysed currently available tools to support decision making or patient understanding in the treatment of acute ischaemic stroke with intravenous thrombolysis, as an exemplar to provide clinicians/researchers with practical guidance on development, evaluation and implementation of such tools for other preference-sensitive treatment options/decisions in different clinical contexts. METHODS: Tools were identified from bibliographic databases, Internet searches and a survey of UK and North American stroke networks. Two reviewers critically analysed tools to establish: information on benefits/risks of thrombolysis included in tools, and the methods used to convey probabilistic information (verbal descriptors, numerical and graphical); adherence to guidance on presenting outcome probabilities (IPDASi probabilities items) and information content (Picker Institute Checklist); readability (Fog Index); and the extent that tools had comprehensive development processes. RESULTS: Nine tools of 26 identified included information on a full range of benefits/risks of thrombolysis. Verbal descriptors, frequencies and percentages were used to convey probabilistic information in 20, 19 and 18 tools respectively, whilst nine used graphical methods. Shortcomings in presentation of outcome probabilities (e.g. omitting outcomes without treatment) were identified. Patient information tools had an aggregate median Fog index score of 10. None of the tools had comprehensive development processes. CONCLUSIONS: Tools to support decision making or patient understanding in the treatment of acute stroke with thrombolysis have been sub-optimally developed. Development of tools should utilise mixed methods and strategies to meaningfully involve clinicians, patients and their relatives in an iterative design process; include evidence-based methods to augment interpretability of textual and probabilistic information (e.g. graphical displays showing natural frequencies) on the full range of outcome states associated with available options; and address patients with different levels of health literacy. Implementation of tools will be enhanced when mechanisms are in place to periodically assess the relevance of tools and where necessary, update the mode of delivery, form and information content.

Life course of retrospective harmonization initiatives: key elements to consider.

Optimizing research on the developmental origins of health and disease (DOHaD) involves implementing initiatives maximizing the use of the available cohort study data; achieving sufficient statistical power to support subgroup analysis; and using participant data presenting adequate follow-up and exposure heterogeneity. It also involves being able to undertake comparison, cross-validation, or replication across data sets. To answer these requirements, cohort study data need to be findable, accessible, interoperable, and reusable (FAIR), and more particularly, it often needs to be harmonized. Harmonization is required to achieve or improve comparability of the putatively equivalent measures collected by different studies on different individuals. Although the characteristics of the research initiatives generating and using harmonized data vary extensively, all are confronted by similar issues. Having to collate, understand, process, host, and co-analyze data from individual cohort studies is particularly challenging. The scientific success and timely management of projects can be facilitated by an ensemble of factors. The current document provides an overview of the 'life course' of research projects requiring harmonization of existing data and highlights key elements to be considered from the inception to the end of the project.

Evidence from the scene: paramedic perspectives on involvement in out-of-hospital research.

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.

Realizing the promise of population biobanks: a new model for translation.

The promise of science lies in expectations of its benefits to societies and is matched by expectations of the realisation of the significant public investment in that science. In this paper, we undertake a methodological analysis of the science of biobanking and a sociological analysis of translational research in relation to biobanking. Part of global and local endeavours to translate raw biomedical evidence into practice, biobanks aim to provide a platform for generating new scientific knowledge to inform development of new policies, systems and interventions to enhance the public's health. Effectively translating scientific knowledge into routine practice, however, involves more than good science. Although biobanks undoubtedly provide a fundamental resource for both clinical and public health practice, their potentiating ontology--that their outputs are perpetually a promise of scientific knowledge generation--renders translation rather less straightforward than drug discovery and treatment implementation. Biobanking science, therefore, provides a perfect counterpoint against which to test the bounds of translational research. We argue that translational research is a contextual and cumulative process: one that is necessarily dynamic and interactive and involves multiple actors. We propose a new multidimensional model of translational research which enables us to imagine a new paradigm: one that takes us from bench to bedside to backyard and beyond, that is, attentive to the social and political context of translational science, and is cognisant of all the players in that process be they researchers, health professionals, policy makers, industry representatives, members of the public or research participants, amongst others.

From genomic databases to translation: a call to action.

The rapid rise of international collaborative science has enabled access to genomic data. In this article, it is argued that to move beyond mapping genomic variation to understanding its role in complex disease aetiology and treatment will require extending data sharing for the purposes of clinical research translation and implementation.

Beyond trust: Amplifying unheard voices on concerns about harm resulting from health data-sharing.

Background: The point of care in many health systems is increasingly a point of health data generation, data which may be shared and used in a variety of ways by a range of different actors. Aim: We set out to gather data about the perspectives on health data-sharing of people living in North East England who have been underrepresented within other public engagement activities and who are marginalized in society. Methods: Multi-site ethnographic fieldwork was carried out in the Teesside region of England over a 6-month period in 2019 as part of a large-scale health data innovation program called Connected Health Cities. Organizations working with marginalized groups were contacted to recruit staff, volunteers, and beneficiaries for participation in qualitative research. The data gathered were analyzed thematically and vignettes constructed to illustrate findings. Results: Previous encounters with health and social care professionals and the broader socio-political contexts of people's lives shape the perspectives of people from marginalized groups about sharing of data from their health records. While many would welcome improved care, the risks to people with socially produced vulnerabilities must be appreciated by those advocating systems that share data for personalized medicine or other forms of data-driven care. Conclusion: Forms of innovation in medicine which rely on greater data-sharing may present risks to groups and individuals with existing vulnerabilities, and advocates of these innovations should address the lack of trustworthiness of those receiving data before asking that people trust new systems to provide health benefits.

Views on genomic research result delivery methods and informed consent: a review.

There has been little discussion of the way genomic research results should be returned and how to obtain informed consent for this. We systematically searched the empirical literature, identifying 63 articles exploring stakeholder perspectives on processes for obtaining informed consent about return of results and/or result delivery. Participants, patients and members of the public generally felt they should choose which results are returned to them and how, ranging from direct (face-to-face, telephone) to indirect (letters, emails, web-based delivery) communication. Professionals identified inadequacies in result delivery processes in the research context. Our findings have important implications for ensuring participants are supported in deciding which results they wish to receive or, if no choice is offered, preparing them for potential research outcomes.

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

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.