Feasibility and ethics of using data from the Scottish newborn blood spot archive for research.

Background: Newborn heel prick blood spots are routinely used to screen for inborn errors of metabolism and life-limiting inherited disorders. The potential value of secondary data from newborn blood spot archives merits ethical consideration and assessment of feasibility for public benefit. Early life exposures and behaviours set health trajectories in childhood and later life. The newborn blood spot is potentially well placed to create an unbiased and cost-effective population-level retrospective birth cohort study. Scotland has retained newborn blood spots for all children born since 1965, around 3 million in total. However, a moratorium on research access is currently in place, pending public consultation. Methods: We conducted a Citizens' Jury as a first step to explore whether research use of newborn blood spots was in the public interest. We also assessed the feasibility and value of extracting research data from dried blood spots for predictive medicine. Results: Jurors delivered an agreed verdict that conditional research access to the newborn blood spots was in the public interest. The Chief Medical Officer for Scotland authorised restricted lifting of the current research moratorium to allow a feasibility study. Newborn blood spots from consented Generation Scotland volunteers were retrieved and their potential for both epidemiological and biological research demonstrated. Conclusions: Through the Citizens' Jury, we have begun to identify under what conditions, if any, should researchers in Scotland be granted access to the archive. Through the feasibility study, we have demonstrated the potential value of research access for health data science and predictive medicine.

Gender stereotypes in UK children and adolescents: Changing patterns of knowledge and endorsement.

Across two studies, we investigated gender stereotype knowledge and endorsement in UK schoolchildren, and their impact on academic subject choice. In Study 1, children aged 9-11 (N = 68) and 13-15 (N = 61) completed a newly developed Gender Attribute scale assessing their knowledge and endorsement of gender stereotypes relating to academic subjects and occupations. Participants demonstrated gender stereotype knowledge and endorsement, although significantly higher knowledge than endorsement scores indicated a level of stereotype rejection. Stereotype knowledge was greater in the older age group, and older girls showed significantly higher levels of stereotype rejection than all other groups. In Study 2, children aged 13-15 (N = 165) completed the Gender Attribute scale and provided information on their school subject choices. Patterns of stereotype knowledge and endorsement followed those of Study 1. Subject choice information showed that boys selected significantly more masculine than feminine subjects, while girls chose a similar proportion of each. Further, boys' level of gender stereotype endorsement predicted their subject choices, while girls' did not. We suggest that in contemporary UK some progress is being made in relation to girls challenging stereotypes that work against them but that more work is needed to encourage boys into female-dominated disciplines.

Public preferences regarding data linkage for research: a discrete choice experiment comparing Scotland and Sweden.

BACKGROUND: There are increasing examples of linking data on healthcare resource use and patient outcomes from different sectors of health and social care systems. Linked data are generally anonymised, meaning in most jurisdictions there are no legal restrictions to their use in research conducted by public or private organisations. Secondary use of anonymised linked data is contentious in some jurisdictions but other jurisdictions are known for their use of linked data. The publics' perceptions of the acceptability of using linked data is likely to depend on a number of factors. This study aimed to quantify the preferences of the public to understand the factors that affected views about types of linked data and its use in two jurisdictions. METHOD: An online discrete choice experiment (DCE) previously conducted in Scotland was adapted and replicated in Sweden. The DCE was designed, comprising five attributes, to elicit the preferences from a representative sample of the public in both jurisdictions. The five attributes (number of levels) were: type of researcher using linked data (four); type of data being linked (four); purpose of the research (three); use of profit from using linked data (four); who oversees the research (four). Each DCE contained 6 choice-sets asking respondents to select their preferred option from two scenarios or state neither were acceptable. Background questions included socio-demographics. DCE data were analysed using conditional and heteroskedastic conditional logit models to create forecasts of acceptability. RESULTS: The study sample comprised members of the public living in Scotland (n = 1004) and Sweden (n = 974). All five attributes were important in driving respondents' choices. Swedish and Scottish preferences were mostly homogenous with the exception of 'who oversees the research using linked data', which had relatively less impact on the choices observed from Scotland. For a defined 'typical' linked data scenario, the probability (on average) of acceptance was 85.7% in Sweden and 82.4% in Scotland. CONCLUSION: This study suggests that the public living in Scotland and Sweden are open to using anonymised linked data in certain scenarios for research purposes but some caution is advisable if the anonymised linked data joins health to non-health data.

Public Preferences regarding Data Linkage for Health Research: A Discrete Choice Experiment.

The potential for data collected in the public and private sector to be linked and used in research has led to increasing interest in public acceptability of data sharing and data linkage. The literature has identified a range of factors that are important for shaping public responses and in particular has noted that public support for research conducted through data linkage or data sharing is contingent on a number of conditions being met. In order to examine the relative importance of these conditions a Discrete Choice Experiment (DCE) was conducted via an online questionnaire among members of Ipsos MORI's online panel in Scotland. The survey was completed by 1,004 respondents. Overall the two most influential factors shaping respondents' preferences are: the type of data being linked; and, how profits are managed and shared. The type of data being linked is roughly twice as important as who the researchers are. There were slight differences across age groups and between genders and slight differences when comparing respondents with and without long term health conditions. The most notable differences between respondents were found when comparing respondents according to employment and working sector. This study provides much needed evidence regarding the relative importance of various conditions which may be essential for securing and sustaining public support for data-linkage in health research. This may be useful for indicating which factors to focus on in future public engagement and has important implications for the design and delivery of research and public engagement activities. The continuously evolving nature of the field means it will be necessary to revisit the key conditions for public support on an ongoing basis and to examine the contexts and circumstances in which these might change.

Public responses to the sharing and linkage of health data for research purposes: a systematic review and thematic synthesis of qualitative studies.

BACKGROUND: The past 10 years have witnessed a significant growth in sharing of health data for secondary uses. Alongside this there has been growing interest in the public acceptability of data sharing and data linkage practices. Public acceptance is recognised as crucial for ensuring the legitimacy of current practices and systems of governance. Given the growing international interest in this area this systematic review and thematic synthesis represents a timely review of current evidence. It highlights the key factors influencing public responses as well as important areas for further research. METHODS: This paper reports a systematic review and thematic synthesis of qualitative studies examining public attitudes towards the sharing or linkage of health data for research purposes. Twenty-five studies were included in the review. The included studies were conducted primarily in the UK and North America, with one study set in Japan, another in Sweden and one in multiple countries. The included studies were conducted between 1999 and 2013 (eight studies selected for inclusion did not report data collection dates). The qualitative methods represented in the studies included focus groups, interviews, deliberative events, dialogue workshops and asynchronous online interviews. RESULTS: Key themes identified across the corpus of studies related to the conditions necessary for public support/acceptability, areas of public concern and implications for future research. The results identify a growing body of evidence pointing towards widespread general-though conditional-support for data linkage and data sharing for research purposes. Whilst a variety of concerns were raised (e.g. relating to confidentiality, individuals' control over their data, uses and abuses of data and potential harms arising) in cases where participants perceived there to be actual or potential public benefits from research and had trust in the individuals or organisations conducting and/or overseeing data linkage/sharing, they were generally supportive. The studies also find current low levels of awareness about existing practices and uses of data. CONCLUSIONS: Whilst the results indicate widespread (conditional) public support for data sharing and linkage for research purposes, a range of concerns exist. In order to ensure public support for future research uses of data greater awareness raising combined with opportunities for public engagement and deliberation are needed. This will be essential for ensuring the legitimacy of future health informatics research and avoiding further public controversy.

Moving from trust to trustworthiness: Experiences of public engagement in the Scottish Health Informatics Programme.

The Scottish Health Informatics Programme (SHIP) was a Scotland-wide research programme exploring ways of collecting, managing and analysing electronic patient records for health research. As part of the SHIP public engagement work stream, a series of eight focus groups and a stakeholder workshop were conducted to explore perceptions of the role, relevance and functions of trust (or trustworthiness) in relation to research practices. The findings demonstrate that the public's relationships of trust and/or mistrust in science and research are not straightforward. This paper aims to move beyond simple descriptions of whether publics trust researchers, or in whom members of the public place their trust, and to explore more fully the bases of public trust/mistrust in science, what trust implies and equally what it means for research/researchers to be trustworthy. This has important implications for public engagement in interdisciplinary projects.

Beyond NIMBYs and NOOMBYs: what can wind farm controversies teach us about public involvement in hospital closures?

BACKGROUND: Many policymakers, researchers and commentators argue that hospital closures are necessary as health systems adapt to new technological and financial contexts, and as population health needs in developed countries shift. However closures are often unpopular with local communities. Previous research has characterised public opposition as an obstacle to change. Public opposition to the siting of wind farms, often described as NIMBYism (Not In My Back Yard), is a useful comparator issue to the perceived NOOMBYism (Not Out Of My Back Yard) of hospital closure protestors. DISCUSSION: The analysis of public attitudes to wind farms has moved from a fairly crude characterisation of the 'attitude-behaviour gap' between publics who support the idea of wind energy, but oppose local wind farms, to empirical, often qualitative, studies of public perspectives. These have emphasised the complexity of public attitudes, and revealed some of the 'rational' concerns which lie beneath protests. Research has also explored processes of community engagement within the wind farm decision-making process, and the crucial role of trust between communities, authorities, and developers. Drawing on what has been learnt from studies of opposition to wind farms, we suggest a range of questions and approaches to explore public perspectives on hospital closure more thoroughly. Understanding the range of public responses to service change is an important first step in resolving the practical dilemma of effecting health system transformation in a democratic fashion.