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ABSTRACT Introduction There is a pressing need to understand and explore the complex experiences and psychosocial support needs of people LWBC‐CM and their informal caregivers, to inform survivorship and supportive care interventions. Methods In‐depth qualitative interviews were conducted with people LWBC‐CM and their informal caregivers in Scotland, invited via primary care. One‐to‐one, face‐to‐face interviews were conducted with informed consent exploring experiences of symptoms, psychosocial support needs and interactions with health services. Interviews were transcribed and analysed using a thematic approach. Results Forty‐one people LWBC‐CM and twenty‐three informal caregivers were interviewed. Four themes were identified: the Physical and Psychological Impact of Cancer and Comorbidity, Dominant Storie—Prioritising Conditions and Making Sense of Illness, Navigating Health Services and Treatments and Caring for People with Complex Health Conditions. Type and severity of conditions mediated people's experiences and daily living. Complex fatigue—fatigue arising from a number of health conditions—dominated symptomology. Participants navigated multiple appointments and complex medication regimes. Patients identified the need for acknowledgement of other chronic conditions and for streamlined care provision. Mutual caring and social isolation were also identified as part of the caring relationship. Conclusions There is a mandate to address the psychosocial support needs of people LWBC‐CM, and their informal carers, given the burden of treatment for cancer survivors with moderate to severe complex conditions as they navigate health services. Patient or Public Contribution A patient representative has been involved in all stages of the study from development of the application through study design, commenting on documentation, analysis of transcripts and writing the manuscript. They are included as an author on the manuscript.

Cunningham-Burley, McCartney, Campbell, Flaig, Orange et al. assess the ethics and potential benefit of secondary data use from newborn blood spot archives for research in a Citizen’s jury that agreed this was in the public interest. They also investigate and demonstrate the feasibility of using the archives for research purposes.

Objectives There were two objectives to this study: 1) to gauge public opinion on the use of Guthrie card-derived blood samples for epidemiological and biological research; and 2) to evaluate the feasibility of recovering meaningful molecular data from these samples. Approach To address the first objective, a 2-day Citizens’ Jury was conducted in partnership with Ipsos MORI, comprising a diverse adult sample in terms of age, sex, working status and social grade (n=20). Jurors were asked whether research access to Guthrie card blood tests would be in the public interest. To address the second objective, DNA methylation (DNAm) was profiled from samples from 58 Generation Scotland participants, whose Guthrie cards had been stored from birth for between 32 and 38 years. Analyses were performed on Guthrie DNAm samples to determine whether previously-reported associations with perinatal maternal smoking behaviours were detectable. Results The Citizens’ Jury yielded an overall positive response towards data sharing for health research. Concerns were raised about data protection and security, control and oversight, and commercial use. The overall verdict was that access to Guthrie card data would be in the public interest, conditional on the purpose of the research, regulated access procedures, ethical oversight and provision of opportunities for participants to opt out. DNAm detection rates from Guthrie samples were lower than from samples stored in tubes. However, it was possible to confirm linkage to the correct individuals in Generation Scotland using DNAm-derived estimates of genotype and sex. A significant association was observed between a DNAm-based score for smoking and perinatal maternal smoking status derived from the baseline Generation Scotland questionnaire. Conclusion We showed that: 1) public support exists for using Guthrie samples in research, conditional on certain safeguards; 2) DNAm can be profiled from cards stored for up to 38 years and can predict maternal smoking behaviour. Guthrie cards are a potentially valuable resource for epidemiological studies and predicting health outcomes.

As precision oncology has evolved, patients and their families have become more involved in efforts to access these treatments via fundraising and campaigning that take place outside of the larger cancer charities. In this paper, we explore the solidarities, networks, and emotional work of the UK-based access advocates, drawing on the stories of nine advocates, which included interviews and content analyses of their social media posts and coverage of their case in news, commentary, and fundraising websites. We consider the emotional and knowledge work of building networks that spanned consumerist and activist agendas, forged individual and collective goals, and orientations toward the public, private, and third sectors as part of securing support and access. Through these various practices, the actors we have studied cultivated personal advantage and solidarities with other patients and advocates, and in so doing engaged in self and collective advocacy alongside and beyond mainstream cancer charities.

Engagement with publics, patients, and stakeholders is an important part of the health research environment today,and different modalities of ‘engaged’ health research have proliferated in recent years. Yet, there is no consensus on what, exactly, ‘engaging’ means, what it should look like, and what the aims, justifications, or motivations for it should be. In this paper, we set out what we see as important, outstanding challenges around the practice and theory of engaging and consider the tensions and possibilities that the diverse landscape of engaging evokes. We examine the roots, present modalities and institutional frameworks that have been erected around engaging, including how they shape and delimit how engagements are framed, enacted, and justified. We inspect the related issue of knowledge production within and through engagements, addressing whether engagements can, or should, be framed as knowledge producing activities. We then unpack the question of how engagements are or could be valued and evaluated, emphasising the plural ways in which ‘value’ can be conceptualised and generated. We conclude by calling for a philosophy of engagements that can capture the diversity of related practices, concepts and justifications around engagements, and account for the plurality of knowledges and value that engagements engender, while remaining flexible and attentive to the structural conditions under which engagements occur. Such philosophy should be a feminist one, informed by feminist epistemological and methodological approaches to equitable modes of research participation, knowledge production, and valuing. Especially, translating feminist tools of reflexivity and positionalityinto the sphere of engagements can enable a synergy of empirical, epistemic and normative considerations in developing accounts of engaging in both theory and praxis. Modestly, here, we hope to carve out the starting points for this work.

Objective To explore the experiences of pregnancy, childbirth, antenatal and postnatal care in women belonging to ethnic minorities and to identify any specific challenges that these women faced during the SARS-CoV-2 pandemic.Design This was a qualitative study using semistructured interviews of pregnant women or those who were 6 weeks postnatal from Black, Asian and minority ethnic backgrounds. The study included 16 women in a predominantly urban Scottish health board area.Results The finding are presented in four themes: ‘communication’, ‘interactions with healthcare professionals’, ‘racism’ and ‘the pandemic effect’. Each theme had relevant subthemes. ‘Communication’ encompassed respect, accent bias, language barrier and cultural dissonance; ‘interactions with healthcare professionals’: continuity of care, empathy, informed decision making and dissonance with other healthcare systems; ‘racism’ was deemed to be institutional, interpersonal or internalised; and ‘the pandemic effect’ consisted of isolation, psychological impact and barriers to access of care.Conclusions This study provides insight into the specific challenges faced by ethnic minority women in pregnancy, which intersect with the unique problems posed by the ongoing SARS-CoV-2 pandemic to potentially widen existing ethnic disparities in maternal outcomes and experiences of maternity care.

Background: Preterm birth is common in twins and accounts for significant mortality and morbidity. There are no effective preventative treatments. Some studies have suggested that, in twin pregnancy complicated by a short cervix, the Arabin pessary, which fits around the cervix and can be inserted as an outpatient procedure, reduces preterm birth and prevents neonatal morbidity. Objective: STOPPIT 2 aimed to evaluate the clinical utility of the Arabin cervical pessary in preventing preterm birth in women with a twin pregnancy and a short cervix. Design: STOPPIT 2 was a pragmatic, open label, multicentre randomised controlled trial with two treatment group – the Arabin pessary plus standard care (intervention) and standard care alone (control). Participants were initially recruited into the screening phase of the study, when cervical length was measured. Women with a measured cervical length of ≤ 35 mm were then recruited into the treatment phase of the study. An economic evaluation considered cost-effectiveness and a qualitative substudy explored the experiences of participants and clinicians. Setting: Antenatal clinics in the UK and elsewhere in Europe. Participants: Women with twin pregnancy at

This paper explores clinicians’ and scientists’ accounts of genomic research in cancer care and the complexities and challenges involved with delivering this work. Contributing to the sociology of (low) expectations, we draw on sociological studies of uncertainty in medicine to explore their accounts of working with uncertainty as part of the management of patient and institutional expectations. We consider their appeals to the importance of modest inquiry and framing of the uncertainties of genomic medicine as normal and at times welcome as they sought to configure professional autonomy and jurisdictions and cultivate an experimental ethos amongst their patients. We argue that these types of uncertainty work [Star, S. L. 1985. “Scientific Work and Uncertainty.” Social Studies of Science 15 (3): 391–427] are a key feature of managing expectations at the intersections of genomic research and clinical care.

BackgroundPreterm-labour-associated preterm birth is a common cause of perinatal mortality and morbidity in twin pregnancy. We aimed to test the hypothesis that the Arabin pessary would reduce preterm-labour-associated preterm birth by 40% or greater in women with a twin pregnancy and a short cervix.Methods and findingsWe conducted an open-label randomised controlled trial in 57 hospital antenatal clinics in the UK and Europe. From 1 April 2015 to 14 February 2019, 2,228 women with a twin pregnancy underwent cervical length screening between 18 weeks 0 days and 20 weeks 6 days of gestation. In total, 503 women with cervical length ≤ 35 mm were randomly assigned to pessary in addition to standard care (n = 250, mean age 32.4 years, mean cervical length 29 mm, with pessary inserted in 230 women [92.0%]) or standard care alone (n = 253, mean age 32.7 years, mean cervical length 30 mm). The pessary was inserted before 21 completed weeks of gestation and removed at between 35 and 36 weeks or before birth if earlier. The primary obstetric outcome, spontaneous onset of labour and birth before 34 weeks 0 days of gestation, was present in 46/250 (18.4%) in the pessary group compared to 52/253 (20.6%) following standard care alone (adjusted odds ratio [aOR] 0.87 [95% CI 0.55-1.38], p = 0.54). The primary neonatal outcome-a composite of any of stillbirth, neonatal death, periventricular leukomalacia, early respiratory morbidity, intraventricular haemorrhage, necrotising enterocolitis, or proven sepsis, from birth to 28 days after the expected date of delivery-was present in 67/500 infants (13.4%) in the pessary group compared to 76/506 (15.0%) following standard care alone (aOR 0.86 [95% CI 0.54-1.36], p = 0.50). The positive and negative likelihood ratios of a short cervix (≤35 mm) to predict preterm birth before 34 weeks were 2.14 and 0.83, respectively. A meta-analysis of data from existing publications (4 studies, 313 women) and from STOPPIT-2 indicated that a cervical pessary does not reduce preterm birth before 34 weeks in women with a short cervix (risk ratio 0.74 [95% CI 0.50-1.11], p = 0.15). No women died in either arm of the study; 4.4% of babies in the Arabin pessary group and 5.5% of babies in the standard treatment group died in utero or in the neonatal period (p = 0.53). Study limitations include lack of power to exclude a smaller than 40% reduction in preterm labour associated preterm birth, and to be conclusive about subgroup analyses.ConclusionsThese results led us to reject our hypothesis that the Arabin pessary would reduce the risk of the primary outcome by 40%. Smaller treatment effects cannot be ruled out.Trial registrationISRCTN Registry ISRCTN 02235181. ClinicalTrials.gov NCT02235181.

The global coronavirus pandemic has clearly demonstrated the great urgency to collect and use patient data effectively to understand, track and manage the spread of Covid-19. The value of patient data in this pandemic is undeniable, however considerations around how - and by whom - such data should be collected, accessed and used, and for what purposes, remain to be fully debated and resolved. Who decides, and how such decisions are made remain unclear. We argue that public engagement and deliberation are essential for good governance and are key to establish and maintain a legitimate social licence for data practices around Covid-19.

Sociological literature has explored how shifts in the point at which individuals may be designated as diseased impact upon experiences of ill health. Research has shown that experiences of being genetically “at risk” are shaped by and shape familial relations, coping strategies, and new forms of biosociality. Less is known about how living with genetic risk is negotiated in the everyday and over time, and the wider forms of identity, communities and care this involves. This article explores these arrangements drawing on online bloggers’ accounts of Familial Adenomatous Polyposis (FAP). We show how accounts of genetic risk co-exist with more palpable experiences of FAP in everyday life, notably the consequences of prophylactic surgeries. We consider how the act of blogging represents but also constitutes everyday experiences of hereditary cancer syndrome as simultaneously ordinary and exceptional, and reflect on the implications of our analysis for understanding experiences of genetic cancer risk.

The ‘digital era’ of informatics and knowledge integration has changed the roles and experiences of patients, research participants and health consumers. No longer figured (merely) as passive recipients of healthcare services or as beneficiaries of top-down biomedical information, individuals are increasingly seen as active contributors in healthcare and research. They are positioned into multiple roles that are experienced simultaneously by those who access and co-produce digital content that can easily be transformed into data. This is contextualised by ‘big data’ technologies that have altered biomedicine, enabling collation and analysis of myriad data from digitised records to personal mobile data. Social media facilitate new formations of communities and knowledge enacted online, while novel kinds of commercial value emerge from digital networks that enable health data commodification. In this paper, we draw from exemplary digital era shifts towards participatory medicine to cast light on the rapprochements between patienthood, participation and consumption, and we explore how these rapprochements are mediated by, and materialise through, the use of participatory digital technologies and big data. We argue that there is a need to use new conceptual tools that account for the multiple roles and experiences of patient–participant–consumers that co-emerge through digital technologies. We must also ethically re-assess the rights and responsibilities of individuals in the digital era, and the implications of digital era changes for the future of biomedicine and healthcare.

This consensus statement reflects the deliberations of an international group of stakeholders with a range of expertise in public involvement and engagement (PI&E) relating to data-intensive health research. It sets out eight key principles to establish a secure role for PI&E in and with the research community internationally and ensure best practice in its execution. Our aim is to promote culture change and societal benefits through ensuring a socially responsible trajectory for innovations in this field. Our key premise is that the public should not be characterised as a problem to be overcome but a key part of the solution to establish socially beneficial data-intensive health research for all.

The commitment of massive resources – financial, social, organisational, and human – drives developments in biomedicine. Fundamental transformations in the generation and application of knowledge are challenging our understandings and experiences of health, illness, and disease as well as the organisation of research and care. Coupled with the accelerated pace of change, it is pressing that we build authentic collaborations across and between the biomedical sciences, humanities and social sciences, and wider society. It is only in this way that we can ask and answer the penetrating questions that will shape improvements in human health now and in the decades ahead. We delineate the need for such commitments across five key areas of human and societal experience that impact on and are impacted by developments in biomedicine: disease; bodies; global movements and institutions; law; and, science-society engagements. Interactions between ideas, researchers, and communities across and within these domains can provide a way into creating the new knowledges, methods, and partnerships we believe are essential if the promises of biomedicine are to be realised.

The digitization of society and academic research endeavours have led to an explosion of interest in the potential uses of population data in research. Alongside this, increasing attention is focussing on the conditions necessary for maintaining a social license for research practices. Previous research has pointed to the importance of demonstrating “public benefits” from research for maintaining public support, yet there has been very little consideration of what the term “public benefits” means or what public expectations of “public benefits” are. In order to address this pressing issue a series of deliberative workshops with members of the public were held across Scotland in May and June 2017. The workshops aimed to engage a cross-section of the Scottish population in in-depth discussions of the ways that the public – or publics – might benefit from data-intensive health research. The findings reported here discuss workshop participants’ understandings and expectations of health research; who they considered to be “the public” that should benefit from health research and; in what ways they felt “the public” should benefit. Workshop participants’ preference was clearly for the widest possible public benefit to be felt by all, but they also acknowledged the value in research aiming to primarily benefit vulnerable groups within society. A key focus of discussions was the extent to which workshop participants were confident that potential public benefits would be realised. A crucial consideration then is the extent to which mechanisms and political support are in place to realise and maximise the public benefits of data-intensive health research.

Introduction Data-intensive health research is a fast moving field in which public involvement and engagement (PI\&E) is essential for developing socially acceptable and ethically robust processes and ensuring a social license for research practices. Nevertheless while some consensus is emerging around the importance of PI\&E, commitments and practices are varied. Objectives and Approach Our objective was to identify a set of principles which would underpin international best practice in data-intensive health research. A one-day consensus workshop was held in Manchester, U.K. in April 2017 and attended by 31 international participants from six countries, with a range of expertise relating to PI&E with data-intensive health research. Participants took part in facilitated discussions to identify key principles and a consensus statement was written to reflect the discussions at the workshop, this developed through iterative drafts on which all workshop participants as well as the four Farr Institute public panels were consulted. Results The consensus statement sets out key principles to establish a secure role for PI&E related to data-intensive health research internationally and to ensure best practice in its execution. While methods used may not vary greatly from PI&E with other types of research, or other policy areas, we have identified a number of particular features of data-intensive health research which make PI&E in this area worthy of special consideration. Often using data from multiple sources without explicit consent, PI&E in data-intensive health research is particularly important for raising awareness, for giving people a voice and for enabling people to participate in processes of research and governance. The consensus statement sets out the key principles to guide future best practice in PI&E with data-intensive health research. Conclusion/Implications The emergence of data intensive health research and the importance of the social contract upon which it relies, demands that we move beyond rhetorical commitments and engage anew with clearly stated principles to build PI\&E into data-intensive health research at all levels.

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. .

Background: The United Kingdom (UK) lags behind other high-income countries in relation to technological innovation in healthcare. In order to inform UK strategy on how to catalyse innovation, we sought to understand what national strategies can help to promote a climate for innovation in healthcare settings by extracting lessons for the UK from international innovators. Methods: We undertook a series of qualitative semi-structured interviews with senior international innovators from a range of health related policy, care/service delivery, commercial and academic backgrounds. Thematic analysis helped to explore how different stakeholder groups could facilitate/inhibit innovation at individual, organisational, and wider societal levels. Results: We conducted 14 interviews and found that a conducive climate for healthcare innovation comprised of national/regional strategies stimulating commercial competition, promoting public/private relationships, and providing central direction (e.g. incentives for adoption and regulation through standards) without being restrictive. Organisational attitudes with a willingness to experiment and to take risks were also seen as important, but a bottom-up approach to innovation, based on the identification of clinical need, was seen as a crucial first step to construct relevant national policies. Conclusions: There is now a need to create mechanisms through which frontline National Health Service staff in relation can raise ideas/concerns and suggest opportunities for improvement, and then build national innovation environments that seek to address these needs. This should be accompanied by creating competitive health technology markets to stimulate a commercial environment that attracts high-quality health information technology experts and innovators working in partnership with staff and patients.

Background: Maternal obesity is associated with high birthweight, obesity and premature mortality in adult offspring, probably as a result of maternal hyperglycaemia and insulin resistance. We present the results of a trial designed to test the hypothesis that metformin will improve insulin sensitivity in obese pregnant women, thereby reducing the incidence of high-birthweight babies. Objective: To determine the efficacy of metformin (up to 2500 mg daily) given to obese pregnant women in reducing the gestational age-, parity- and sex-adjusted birthweight centile of the baby. Design: Double-blind, placebo-controlled, randomised controlled trial with embedded substudies. Setting: Fifteen NHS hospitals in the UK. Participants: Pregnant women aged ≥ 16 years with a singleton fetus and a body mass index of ≥ 30 kg/m2. Intervention: Metformin tablets (or placebo) administered between 12 and 16 weeks’ gestation until delivery of the baby. Main outcome measures: The primary outcome measure was z-score corresponding to the gestational age-, parity- and sex-adjusted birthweight centile of live-born babies delivered at ≥ 24 weeks’ gestation. The main secondary outcome was maternal insulin resistance at 36 weeks’ gestation. Embedded substudies were included to assess the effect of metformin on insulin sensitivity using the hyperinsulinaemic–euglycaemic clamp; endothelial function; maternal and fetal fat distribution using magnetic resonance imaging; placental expression of 11β-hydroxysteroid dehydrogenase types 1 and 2 and glucocorticoid receptor; and myometrial contractility and glycogen storage. Results: We randomised 449 women to either placebo (n = 223) or metformin (n = 226), of whom 434 were included in the final intention-to-treat analysis. Mean birthweight at delivery was 3463 g [standard deviation (SD) 660 g] in the placebo group and 3462 g (SD 548 g) in the metformin group. The estimated effect size of metformin on the primary outcome was non-significant [adjusted mean difference in z-score –0.029, 95% confidence interval (CI) –0.217 to 0.158; p = 0.7597]. There was no evidence of a reduction in the main secondary outcome of homeostatic model assessment – insulin resistance (HOMA-IR) at 36 weeks’ gestation (mean HOMA-IR 5.98 and 6.30 molar units in the placebo and metformin groups, respectively; adjusted mean ratio 0.974, 95% CI 0.865 to 1.097). Metformin had no effect on the combined adverse outcome of miscarriage, termination of pregnancy, stillbirth or neonatal death. Subjects taking metformin demonstrated increased insulin sensitivity [glucose disposal per unit plasma insulin difference between means during high-dose insulin 0.02 mg/kg, 95% CI 0.001 to 0.03 mg/kg (fat-free mass)/minute/µIU/l; p = 0.04] compared with those taking placebo and enhanced endogenous glucose production [difference between means 0.54 mg/kg, 95% CI 0.08 to 1.00 mg/kg (fat-free mass)/minute; p = 0.02]. There were no differences in endothelial function, maternal or fetal body fat distribution, placental expression of 11β-hydroxysteroid dehydrogenase types 1 and 2 and glucocorticoid receptor, or myometrial contractility and glycogen storage. Conclusions: Metformin has no clinically significant effect on birthweight centile in obese pregnant women. Follow-up studies of the children born to participants in the trial are required to determine whether or not there are any longer-term benefits or harms of maternal metformin for offspring weight, fat mass or metabolism. Trial registration: Current Controlled Trials ISRCTN51279843. Funding: This project was funded by the Efficacy and Mechanism Evaluation programme, a Medical Research Council and National Institute for Health Research partnership.

Objective The global research group, DIPLOMATIC (Using eviDence, Implementation science, and a clinical trial PLatform to Optimise MATernal and newborn health in low Income Countries), aims to reduce stillbirths and preterm births and optimise outcomes for babies born preterm. Minimum datasets for routine data collection in healthcare facilities participating in DIPLOMATIC (initially in Malawi) were designed to assist understanding of baseline maternal and neonatal care processes and outcomes, and facilitate evaluation of improvement interventions and pragmatic clinical trials.Design Published and grey literature was reviewed alongside extensive in-country consultation to define relevant clinical best practice guidance, and the existing local data and reporting infrastructure, to identify requirements for the minimum datasets. Data elements were subjected to iterative rounds of consultation with topic experts in Malawi and Scotland, the relevant Malawian professional bodies and the Ministry of Health in Malawi to ensure relevance, validity and feasibility.Setting Antenatal, maternity and specialist neonatal care in Malawi.Results The resulting three minimum datasets cover the maternal and neonatal healthcare journey for antenatal, maternity and specialist neonatal care, with provision for effective linkage of records for mother/baby pairs. They can facilitate consistent, precise recording of relevant outcomes (stillbirths, preterm births, neonatal deaths), risk factors and key care processes.Conclusions Poor quality routine data on care processes and outcomes constrain healthcare system improvement. The datasets developed for implementation in DIPLOMATIC partner facilities reflect, and hence support delivery of, internationally agreed best practice for maternal and newborn care in low-income settings. Informed by extensive consultation, they are designed to integrate with existing local data infrastructure and reporting as well as meeting research data needs. This work provides a transferable example of strengthening data infrastructure to underpin a learning healthcare system approach in low-income settings.DIPLOMATIC is funded by the UK National Institute for Health Research.