Gestational age and hospital admission costs from birth to childhood: a population-based record linkage study in England.

OBJECTIVE: To examine the association between gestational age at birth and hospital admission costs from birth to 8 years of age. DESIGN: Population-based, record linkage, cohort study in England. SETTING: National Health Service (NHS) hospitals in England, UK. PARTICIPANTS: 1 018 136 live, singleton births in NHS hospitals in England between 1 January 2005 and 31 December 2006. MAIN OUTCOME MEASURES: Hospital admission costs from birth to age 8 years, estimated by gestational age at birth (<28, 28-29, 30-31, 32, 33, 34, 35, 36, 37, 38, 39, 40, 41 and 42 weeks). RESULTS: Both birth admission and subsequent admission hospital costs decreased with increasing gestational age at birth. Differences in hospital admission costs between gestational age groups diminished with increasing age, particularly after the first 2 years following birth. Children born extremely preterm (<28 weeks) and very preterm (28-31 weeks) still had higher average hospital admission costs (£699 (95% CI £419 to £919) for <28 weeks; £434 (95% CI £305 to £563) for 28-31 weeks) during the eighth year of life compared with children born at 40 weeks (£109, 95% CI £104 to £114). Children born extremely preterm had the highest 8-year cumulative hospital admission costs per child (£80 559 (95% CI £79 238 to £82 019)), a large proportion of which was incurred during the first year after birth (£71 997 (95% CI £70 866 to £73 097)). CONCLUSIONS: The association between gestational age at birth and hospital admission costs persists into mid-childhood. The study results provide a useful costing resource for future economic evaluations focusing on preventive and treatment strategies for babies born preterm.

Associations between gestational age at birth and infection-related hospital admission rates during childhood in England: Population-based record linkage study.

BACKGROUND: Children born preterm (<37 completed weeks' gestation) have a higher risk of infection-related morbidity than those born at term. However, few large, population-based studies have investigated the risk of infection in childhood across the full spectrum of gestational age. The objectives of this study were to explore the association between gestational age at birth and infection-related hospital admissions up to the age of 10 years, how infection-related hospital admission rates change throughout childhood, and whether being born small for gestational age (SGA) modifies this relationship. METHODS AND FINDINGS: Using a population-based, record-linkage cohort study design, birth registrations, birth notifications and hospital admissions were linked using a deterministic algorithm. The study population included all live, singleton births occurring in NHS hospitals in England from January 2005 to December 2006 (n = 1,018,136). The primary outcome was all infection-related inpatient hospital admissions from birth to 10 years of age, death or study end (March 2015). The secondary outcome was the type of infection-related hospital admission, grouped into broad categories. Generalised estimating equations were used to estimate adjusted rate ratios (aRRs) with 95% confidence intervals (CIs) for each gestational age category (<28, 28-29, 30-31, 32, 33, 34, 35, 36, 37, 38, 39, 40, 41 and 42 weeks) and the models were repeated by age at admission (<1, 1-2, 3-4, 5-6, and 7-10 years). An interaction term was included in the model to test whether SGA status modified the relationship between gestational age and infection-related hospital admissions. Gestational age was strongly associated with rates of infection-related hospital admissions throughout childhood. Whilst the relationship attenuated over time, at 7-10 years of age those born before 40 weeks gestation were still significantly higher in comparison to those born at 40 weeks. Children born <28 weeks had an aRR of 6.53 (5.91-7.22) during infancy, declining to 3.16 (2.50-3.99) at ages 7-10 years, in comparison to those born at 40 weeks; whilst in children born at 38 weeks, the aRRs were 1·24 (1.21-1.27) and 1·18 (1.13-1.23), during infancy and aged 7-10 years, respectively. SGA status modified the effect of gestational age (interaction P<0.0001), with the highest rate among the children born at <28 weeks and SGA. Finally, study findings indicated that the associations with gestational age varied by subgroup of infection. Whilst upper respiratory tract infections were the most common type of infection experienced by children in this cohort, lower respiratory tract infections (LRTIs) (<28 weeks, aRR = 10.61(9.55-11.79)) and invasive bacterial infections (<28 weeks, aRR = 6.02 (4.56-7.95)) were the most strongly associated with gestational age at birth. Of LRTIs experienced, bronchiolitis (<28 weeks, aRR = 11.86 (10.20-13.80)), and pneumonia (<28 weeks, aRR = 9.49 (7.95-11.32)) were the most common causes. CONCLUSIONS: Gestational age at birth was strongly associated with rates of infection-related hospital admissions during childhood and even children born a few weeks early remained at higher risk at 7-10 years of age. There was variation between clinical subgroups in the strength of relationships with gestational age. Effective infection prevention strategies should include focus on reducing the number and severity of LRTIs during early childhood.

Gestational age and hospital admissions during childhood: population based, record linkage study in England (TIGAR study).

OBJECTIVE: To examine the association between gestational age at birth and hospital admissions to age 10 years and how admission rates change throughout childhood. DESIGN: Population based, record linkage, cohort study in England. SETTING: NHS hospitals in England, United Kingdom. PARTICIPANTS: 1 018 136 live, singleton births in NHS hospitals in England between January 2005 and December 2006. MAIN OUTCOME MEASURES: Primary outcome was all inpatient hospital admissions from birth to age 10, death, or study end (March 2015); secondary outcome was the main cause of admission, which was defined as the World Health Organization's first international classification of diseases, version 10 (ICD-10) code within each hospital admission record. RESULTS: 1 315 338 admissions occurred between 1 January 2005 and 31 March 2015, and 831 729 (63%) were emergency admissions. 525 039 (52%) of 1 018 136 children were admitted to hospital at least once during the study period. Hospital admissions during childhood were strongly associated with gestational age at birth (<28, 28-29, 30-31, 32, 33, 34, 35, 36, 37, 38, 39, 40, 41, and 42 weeks). In comparison with children born at full term (40 weeks' gestation), those born extremely preterm (<28 weeks) had the highest rate of hospital admission throughout childhood (adjusted rate ratio 4.92, 95% confidence interval 4.58 to 5.30). Even children born at 38 weeks had a higher rate of hospital admission throughout childhood (1.19, 1.16 to 1.22). The association between gestational age and hospital admission decreased with increasing age (interaction P<0.001). Children born earlier than 28 weeks had an adjusted rate ratio of 6.34 (95% confidence interval 5.80 to 6.85) at age less than 1 year, declining to 3.28 (2.82 to 3.82) at ages 7-10, in comparison with those born full term; whereas in children born at 38 weeks, the adjusted rate ratios were 1.29 (1.27 to 1.31) and 1.16 (1.13 to 1.19), during infancy and ages 7-10, respectively. Infection was the main cause of excess hospital admissions at all ages, but particularly during infancy. Respiratory and gastrointestinal conditions also accounted for a large proportion of admissions during the first two years of life. CONCLUSIONS: The association between gestational age and hospital admission rates decreased with age, but an excess risk remained throughout childhood, even among children born at 38 and 39 weeks of gestation. Strategies aimed at the prevention and management of childhood infections should target children born preterm and those born a few weeks early.

Linkage of maternity hospital episode statistics birth records to birth registration and notification records for births in England 2005-2006: quality assurance of linkage.

OBJECTIVES: The objectives of this study were to describe the methods used to assess the quality of linkage between records of babies' birth registration and hospital birth records, and to evaluate the potential bias that may be introduced because of these methods. DESIGN/SETTING: Data from the civil registration and the notification of births previously linked by the Office for National Statistics (ONS) had been further linked to birth records from the Hospital Episode Statistics (HES) for babies born in England. We developed a deterministic, six-stage algorithm to assess the quality of this linkage. PARTICIPANTS: All 1 170 790 live, singleton births, occurring in National Health Service hospitals in England between 1 January 2005 and 31 December 2006. PRIMARY OUTCOME MEASURE: The primary outcome was the number of successful links between ONS birth records and HES birth records. Rates of successful linkage were calculated for the cohort and the characteristics associated with unsuccessful linkage were identified. RESULTS: Approximately 92% (1 074 572) of the birth registration records were successfully linked with a HES birth record. Data quality and completeness were somewhat poorer in HES birth records compared with linked birth registration and birth notification records. The quality assurance algorithms identified 1456 incorrect linkages (<1%). Compared with the linked dataset, birth records were more likely to be unlinked if babies were of white ethnic origin; born to unmarried mothers; born in East England, London, North West England or the West Midlands; or born in March. CONCLUSIONS: It is possible to link administrative datasets to create large cohorts, allowing researchers to explore important questions about exposures and childhood outcomes. Missing data, coding errors and inconsistencies mean it is important that the quality of linkage is assessed prior to analysis.

Landscape of Participant-Centric Initiatives for Medical Research in the United States, the United Kingdom, and Japan: Scoping Review.

BACKGROUND: Information and communication technology (ICT) has made remarkable progress in recent years and is being increasingly applied to medical research. This technology has the potential to facilitate the active involvement of research participants. Digital platforms that enable participants to be involved in the research process are called participant-centric initiatives (PCIs). Several PCIs have been reported in the literature, but no scoping reviews have been carried out. Moreover, detailed methods and features to aid in developing a clear definition of PCIs have not been sufficiently elucidated to date. OBJECTIVE: The objective of this scoping review is to describe the recent trends in, and features of, PCIs across the United States, the United Kingdom, and Japan. METHODS: We applied a methodology suggested by Levac et al to conduct this scoping review. We searched electronic databases-MEDLINE (Medical Literature Analysis and Retrieval System Online), Embase (Excerpta Medica Database), CINAHL (Cumulative Index of Nursing and Allied Health Literature), PsycINFO, and Ichushi-Web-and sources of grey literature, as well as internet search engines-Google and Bing. We hand-searched through key journals and reference lists of the relevant articles. Medical research using ICT was eligible for inclusion if there was a description of the active involvement of the participants. RESULTS: Ultimately, 21 PCIs were identified that have implemented practical methods and modes of various communication activities, such as patient forums and use of social media, in the field of medical research. Various methods of decision making that enable participants to become involved in setting the agenda were also evident. CONCLUSIONS: This scoping review is the first study to analyze the detailed features of PCIs and how they are being implemented. By clarifying the modes and methods of various forms of communication and decision making with patients, this review contributes to a better understanding of patient-centric involvement, which can be facilitated by PCIs. INTERNATIONAL REGISTERED REPORT IDENTIFIER (IRRID): RR2-10.2196/resprot.7407.

Motivations for data sharing-views of research participants from four European countries: A DIRECT study.

The purpose of this study was to explore and compare different countries in what motivated research participants' decisions whether to share their de-identified data. We investigated European DIRECT (Diabetes Research on Patient Stratification) research project participants' desire for control over sharing different types of their de-identified data, and with who data could be shared in the future after the project ends. A cross-sectional survey was disseminated among DIRECT project participants. The results found that there was a significant association between country and attitudes towards advancing research, protecting privacy, and beliefs about risks and benefits to sharing data. When given the choice to have control, some participants (<50% overall) indicated that having control over what data is shared and with whom was important; and control over what data types are shared was less important than respondents deciding who data are shared with. Danish respondents indicated higher odds of desire to control data types shared, and Dutch respondents showed higher odds of desire to control who data will be shared with. Overall, what research participants expect in terms of control over data sharing needs to be considered and aligned with sharing for future research and re-use of data. Our findings show that even with de-identified data, respondents prioritise privacy above all else. This study argues to move research participants from passive participation in biomedical research to considering their opinions about data sharing and control of de-identified biomedical data.

Sharing data for future research-engaging participants' views about data governance beyond the original project: a DIRECT Study.

PURPOSE: Biomedical data governance strategies should ensure that data are collected, stored, and used ethically and lawfully. However, research participants' preferences for how data should be governed is least studied. The Diabetes Research on Patient Stratification (DIRECT) project collected substantial amounts of health and genetic information from patients at risk of, and with type II diabetes. We conducted a survey to understand participants' future data governance preferences. Results will inform the postproject data governance strategy. METHODS: A survey was distributed in Denmark, Sweden, The Netherlands, and the United Kingdom. RESULTS: In total 855 surveys were returned. Ninety-seven percent were supportive of sharing data postproject, and 90% were happy to share data with universities, and 56% with commercial companies. The top three priorities for data sharing were highly secure database, DIRECT researchers to monitor data used by other researchers, and researchers cannot identify participants. Respondents frequently suggested that a postproject Data Access Committee should involve a DIRECT researcher, diabetes clinician, patient representative, and a DIRECT participant. CONCLUSION: Preferences of how data should be governed, and what data could be shared and with whom varied between countries. Researchers are considered as key custodians of participant data. Engaging participants aids in designing governance to support their choices.

Do Maternal Dietary Antioxidants Modify the Relationship Between Binge Drinking and Small for Gestational Age? Findings from a Longitudinal Cohort Study.

BACKGROUND: Vitamin C, vitamin E, and carotenoids are potent dietary antioxidants that have been shown to attenuate ethanol-induced harm in animal models of fetal alcohol spectrum disorders. A diet low in antioxidant-rich foods may induce a state of oxidative stress in the context of maternal alcohol consumption during pregnancy, potentially causing growth restriction in the developing fetus. METHODS: We conducted a secondary analysis of a longitudinal U.K. birth cohort. The sample comprised 9,699 women and their babies in Avon, U.K., with an estimated delivery date between April 1, 1991 and December 31, 1992. Alcohol consumption data were self-reported at 18 weeks' gestation via a postal questionnaire. Women reported any binge drinking (≥4 U.K. units/occasion) during the past month. Dietary data were self-reported at 32 weeks' gestation using a food frequency questionnaire. Estimated intakes of vitamins C and E and carotenoids were categorized into quartiles. Logistic regression models with interaction terms were used to investigate relationships between maternal binge drinking, dietary antioxidants, and fetal growth. Models were adjusted for maternal sociodemographic and lifestyle characteristics. Small for gestational age (SGA; <10th percentile) was defined using customized birth centiles. RESULTS: In the unadjusted models, binge drinking was associated with higher risk of SGA birth (odds ratio [OR] 1.38, 95% confidence interval [CI] 1.10, 1.72, p = 0.005), and higher maternal intakes of vitamin C (OR = 0.90, 95% CI 0.84, 0.96, p = 0.002) and vitamin E (OR = 0.90, 95% CI 0.84, 0.95, p < 0.0001) were associated with lower risk of SGA birth. However, addition of potentially confounding variables attenuated these relationships. Likelihood ratio tests indicated that interaction terms were not significant for vitamin C (p = 0.116), vitamin E (p = 0.059), or carotenoid intakes (p = 0.174). CONCLUSIONS: There was no evidence of maternal intake of dietary antioxidants modifying the relationship between maternal binge drinking and SGA birth.

Making the most of the waiting room: Electronic patient engagement, a mixed methods study.

OBJECTIVE: The purpose of this study was to explore whether patients with musculoskeletal conditions would agree to use digital technologies to learn about research registries and make a decision about signing up whilst in the clinic waiting room. METHODS: Patients were recruited from four hospital clinics across Oxfordshire. We used an explanatory mixed methods design with two sequential phases comprising an exploratory, cross-sectional questionnaire (n = 84), followed by focus group interviews (n = 8) to provide context for the findings from the questionnaire. Multivariate ordinal logistic regression models were used to explore relationships between patient preferences and characteristics. Thematic analysis was used to understand the reasons for patient preferences regarding digital technologies and research registries. RESULTS: As participants' age increased, they were more likely to report a preference for face-to-face recruitment methods compared to those using digital technologies. Findings from the focus groups indicated this was primarily due to a fear of technology and physical limitations associated with a patient's condition. Patients also reported a preference for making a decision about signing up at a later date, which was attributed to patients feeling distracted whilst in the waiting room due to anxieties related to their upcoming appointment. CONCLUSIONS: Many patients with musculoskeletal conditions in the UK may be interested in learning about opportunities to participate in research whilst using digital technologies within the waiting room. The results suggest the need for choice regarding the presentation and format of information and whether it can be accessed at a later date at home.

Participant-Centric Initiatives and Medical Research: Scoping Review Protocol.

BACKGROUND: Significant advances in digital technologies have meant that health care data can be collected, stored, transferred, and analyzed for research purposes more easily than ever before. Participant-centric initiatives (PCI) are defined as "tools, programs, and projects that empower participants to engage in the research process" using digital technologies and have the potential to provide a number of benefits to both participants and researchers, including the promotion of public trust in medical research, improved quality of research, increased recruitment and retention, and improved health care delivery. OBJECTIVE: The main objective of this scoping review is to describe the extent and range of PCIs across the United Kingdom, United States, and Japan that are designed to facilitate medical research. METHODS: The methodological framework described by Levac et al will be applied to this scoping review. We will search electronic databases (MEDLINE, EMBASE, PsychINFO, Cumulative Index to Nursing, and Allied Health Literature and CiNii), grey literature sources, Internet search engines (Google and Bing), and hand search key journals and reference lists of relevant articles. All digital tools and programs will be eligible for inclusion if there is a description of key features and functions that fall within the parameters of a PCI. Only those that play a role in medical research will be included. RESULTS: Preliminary searches conducted in MEDLINE and EMBASE retrieved 1820 and 2322 results, respectively. The scoping review will be completed by January 2018. CONCLUSIONS: The scoping review will be the first to map the extent and range of PCIs currently available across the United Kingdom, United States, and Japan, and will be the first review to contribute to a better understanding of what PCIs patients may benefit from. Researchers and practitioners will be able to use information in this review as a guide for patients and also as a guide for the development of future tools and programs. The results will be disseminated through a peer-reviewed publication and conference presentations.

Exploration of dietary patterns and alcohol consumption in pregnant women in the UK: A mixed methods study.

BACKGROUND: fetal Alcohol Spectrum Disorders is a term used to describe a range of physical, cognitive and behavioural deficits in the offspring of women who drank alcohol during pregnancy. A growing body of evidence suggests alcohol consumption in the presence of poor maternal nutrition may increase the risk of harm to the developing fetus. OBJECTIVE: to investigate relationships between maternal dietary patterns and alcohol consumption, and explore which factors influence women's decisions about what to eat and drink during pregnancy. DESIGN: a mixed methods study comprising a questionnaire (paper-based and online) and semi-structured, in-depth interviews with a sub-sample of women who completed the questionnaire. PARTICIPANTS: women were eligible for inclusion if they were ≥16 years of age, pregnant and living in the UK and were recruited through antenatal clinics, specialist substance misuse antenatal clinics or via social media platforms; 350 women completed a questionnaire and a sub-sample of 6 women participated in an interview. METHODS: the questionnaire comprised the Alcohol Use Disorders Identification Test Consumption to measure alcohol consumption patterns and a Food Frequency Questionnaire to measure dietary intake. Dietary pattern analysis was conducted using Principle Components Analysis and linear regression models were used to explore relationships between dietary pattern scores and alcohol consumption. Analyses were adjusted for socio-demographic and lifestyle characteristics. Semi-structured, in-depth interviews were conducted face-to-face and analysed thematically. FINDINGS: two key dietary patterns were derived. Women who reported frequent alcohol consumption before and during pregnancy were more likely to adhere to the 'Prudent' dietary pattern compared to those who abstained. No relationships were observed between alcohol consumption and adherence to the 'Cafeteria' dietary pattern. Six key themes were identified through the qualitative analysis: (1) pregnancy as a time to review behaviour; (2) listen to your body - it will tell you what you need; (3) treats are still important - on special occasions; (4) social and cultural expectations constrain behaviour; (5) inconsistent or ambiguous information creates uncertainty; and 6) confidence increases following a successful pregnancy. CONCLUSIONS: those who drink low levels of alcohol during pregnancy may have better quality diets compared to women who report no alcohol consumption. The reasons for this are complex and influenced by social context and previous pregnancy experience, which should be considered when healthcare professionals provide advice during this period.

Dietary Patterns and Alcohol Consumption During Pregnancy: Secondary Analysis of Avon Longitudinal Study of Parents and Children.

BACKGROUND: Large general population surveys show that heavy regular and episodic alcohol consumption are associated with lower intakes of fruits and vegetables, and higher intakes of processed and fried meat. This is of particular concern regarding pregnant women, as both alcohol intake and inadequate maternal nutrition are independently associated with adverse fetal outcomes. The current study aimed to determine associations between maternal dietary patterns and alcohol consumption during pregnancy. METHODS: Women were participating in the Avon Longitudinal Study of Parents and Children, and provided details of alcohol consumption at 18 weeks' gestation and diet at 32 weeks' gestation (n = 9,839). Dietary patterns were derived from the food frequency questionnaire data using principal components analysis. Associations between alcohol consumption and dietary patterns were determined using multiple linear regression, adjusted for various sociodemographic and lifestyle factors. RESULTS: After adjustment, drinking ≥1 unit/d during the first trimester (ß = 0.23 [95% CI: 0.08, 0.38]; p = 0.002) and binge drinking (≥4 units in 1 day) during the first half of pregnancy (ß = 0.14 [95% CI: 0.07, 0.21]; p < 0.0001) were associated with greater adherence to the "Processed" dietary pattern (high intakes of processed meat and low intakes of fruit and vegetables). Light-to-moderate alcohol consumption (≤1 drink/d) during the first trimester was associated with greater adherence to the "Health conscious" dietary pattern (high intakes of fruit, vegetables, whole grains, and fish) (ß = 0.09 [95% CI: 0.04, 0.14]; p < 0.0001). CONCLUSIONS: Two important components of health behavior during pregnancy appear to be related: greater consumption of processed foods associated with heavier alcohol consumption, and healthier dietary choices associated with light-to-moderate alcohol intake. Potential synergistic effects of these behaviors may have implications for maternal and fetal health and warrant further investigation. A more holistic approach to addressing health behaviors in women of reproductive age is required.

Using digital technologies to engage with medical research: views of myotonic dystrophy patients in Japan.

BACKGROUND: As in other countries, the traditional doctor-patient relationship in the Japanese healthcare system has often been characterised as being of a paternalistic nature. However, in recent years there has been a gradual shift towards a more participatory-patient model in Japan. With advances in technology, the possibility to use digital technologies to improve patient interactions is growing and is in line with changing attitudes in the medical profession and society within Japan and elsewhere. The implementation of an online patient engagement platform is being considered by the Myotonic Dystrophy Registry of Japan. The aim of this exploratory study was to understand patients' views and attitudes to using digital tools in patient registries and engagement with medical research in Japan, prior to implementation of the digital platform. METHODS: We conducted an exploratory, cross-sectional, self-completed questionnaire with a sample of myotonic dystrophy (MD) patients attending an Open Day at Osaka University, Japan. Patients were eligible for inclusion if they were 18 years or older, and were diagnosed with MD. RESULTS: A total of 68 patients and family members attended the Open Day and were invited to participate in the survey. Of those, 59 % submitted a completed questionnaire (n = 40). The survey showed that the majority of patients felt that they were not receiving the information they wanted from their clinicians, which included recent medical research findings and opportunities to participate in clinical trials, and 88 % of patients indicated they would be willing to engage with digital technologies to receive relevant medical information. Patients also expressed an interest in having control over when and how they received this information, as well as being informed of how their data is used and shared with other researchers. CONCLUSION: Overall, the findings from this study suggest that there is scope to develop a digital platform to engage with patients so that they can receive information about medical care and research opportunities. While this study group is a small, self-selecting population, who suffer from a particular condition, the results suggest that there are interested populations within Japan that would appreciate enhanced communication and interaction with healthcare teams.

Challenges and opportunities for ELSI early career researchers.

BACKGROUND: Over the past 25 years, there has been growing recognition of the importance of studying the Ethical, Legal and Social Implications (ELSI) of genetic and genomic research. A large investment into ELSI research from the National Institutes of Health (NIH) Human Genomic Project budget in 1990 stimulated the growth of this emerging field; ELSI research has continued to develop and is starting to emerge as a field in its own right. The evolving subject matter of ELSI research continues to raise new research questions as well as prompt re-evaluation of earlier work and a growing number of scholars working in this area now identify themselves as ELSI scholars rather than with a particular discipline. MAIN TEXT: Due to the international and interdisciplinary nature of ELSI research, scholars can often find themselves isolated from disciplinary or regionally situated support structures. We conducted a workshop with Early Career Researchers (ECRs) in Oxford, UK, and this paper discusses some of the particular challenges that were highlighted. While ELSI ECRs may face many of the universal challenges faced by ECRs, we argue that a number of challenges are either unique or exacerbated in the case of ELSI ECRs and discuss some of the reasons as to why this may be the case. We identify some of the most pressing issues for ELSI ECRs as: interdisciplinary angst and expertise, isolation from traditional support structures, limited resources and funding opportunities, and uncertainty regarding how research contributions will be measured. We discuss the potential opportunity to use web 2.0 technologies to transform academic support structures and address some of the challenges faced by ELSI ECRs, by helping to facilitate mentoring and support, access to resources and new accreditation metrics. CONCLUSION: As our field develops it is crucial for the ELSI community to continue looking forward to identify how emerging digital solutions can be used to facilitate the international and interdisciplinary research we perform, and to offer support for those embarking on, progressing through, and transitioning into an ELSI research career.

Citizen science or scientific citizenship? Disentangling the uses of public engagement rhetoric in national research initiatives.

BACKGROUND: The language of "participant-driven research," "crowdsourcing" and "citizen science" is increasingly being used to encourage the public to become involved in research ventures as both subjects and scientists. Originally, these labels were invoked by volunteer research efforts propelled by amateurs outside of traditional research institutions and aimed at appealing to those looking for more "democratic," "patient-centric," or "lay" alternatives to the professional science establishment. As mainstream translational biomedical research requires increasingly larger participant pools, however, corporate, academic and governmental research programs are embracing this populist rhetoric to encourage wider public participation. DISCUSSION: We examine the ethical and social implications of this recruitment strategy. We begin by surveying examples of "citizen science" outside of biomedicine, as paradigmatic of the aspirations this democratizing rhetoric was originally meant to embody. Next, we discuss the ways these aspirations become articulated in the biomedical context, with a view to drawing out the multiple and potentially conflicting meanings of "public engagement" when citizens are also the subjects of the science. We then illustrate two uses of public engagement rhetoric to gain public support for national biomedical research efforts: its post-hoc use in the "care.data" project of the National Health Service in England, and its proactive uses in the "Precision Medicine Initiative" of the United States White House. These examples will serve as the basis for a normative analysis, discussing the potential ethical and social ramifications of this rhetoric. We pay particular attention to the implications of government strategies that cultivate the idea that members of the public have a civic duty to participate in government-sponsored research initiatives. We argue that such initiatives should draw from policy frameworks that support normative analysis of the role of citizenry. And, we conclude it is imperative to make visible and clear the full spectrum of meanings of "citizen science," the contexts in which it is used, and its demands with respect to participation, engagement, and governance.