The inclusion of qualitative studies in systematic reviews poses methodological challenges. This paper presents worked examples of two methods of data synthesis , used in relation to one review, with the aim of enabling researchers to consider the strength of different approaches.A systematic review of lay perspectives of infant size and growth was conducted, locating 19 studies . The data extracted from these were synthesised using both a textual narrative and a thematic synthesis.The processes of both methods are presented, showing a stepwise progression to the final synthesis. Both methods led us to similar conclusions about lay views toward infant size and growth. Differences between methods lie in the way they dealt with study quality and heterogeneity.On the basis of the work reported here, we consider textual narrative and thematic synthesis have strengths and weaknesses in relation to different research questions. Thematic synthesis holds most potential for hypothesis generation, but may obscure heterogeneity and quality appraisal. Textual narrative synthesis is better able to describe the scope of existing research and account for the strength of evidence, but is less good at identifying commonality. The inclusion of qualitative data in systematic reviews is an area of ongoing methodological development -3, with Innovative methods are often developed during the course of research, but in general, papers report methods only briefly. As a result, the material that could inform learning is more often to be found in filing cabinets than in journals. In this paper we aim to distinguish between "the trivial and non-trivial points of divergence" p.31  by proviA systematic review of lay views about infant size and growth was undertaken as part of a series of interlinked reviews examining the evidence for associations between early growth and a number of later outcomes. The systematic review of views included both qualitative and quantitative studies.Study methods and findings are reported in greater detail elsewhere -13. StanTwo researchers independently extracted findings by interrogating each study using the following questions developed from the aims of the review:1. What is healthy growth/size?2. How important is growth/size to participants?3. What concepts are used to define healthy growth/size?4. How do participants assess growth/size?5. Where does growth lie among priorities for child health?6. What information influences views/behaviour?7. Who influences views/behaviour?Directly reported participant data  and author interpretations were recorded separately, to retain the richness or 'thickness' of the contributing data. 'Thickness' in this context refers to the kinds of relatively detailed descriptions and contextual material which help the reader to make judgements about the trustworthiness of the data, particularly when applying it to different contexts ,16. StudTwo methods were proposed for synthesis of findings, textual narrative and thematic, both of which the advisory group agreed were appropriate to our needs. The first, the textual narrative approach, involves a commentary reporting on study characteristics, context, quality, and findings, using the scope, differences and similarities among studies were used to draw conclusions across the studies, whilst the second, the thematic approach, groups data into the themes. Given the relatively small number of studies located, it was feasible to test both methods. Findings from the review are provided briefly for illustration, but the focus of this paper is on the process of synthesis and a comparison of methods used. The two reviews ran in tandem, as the thematic review needed time for response and comparison between reviewers.Factors identified by the research team from the research literature as likely to affect views on infant growth were used to define a number of sub-groups. These were:1. Relationship between participant and infant 2. Weight status of participant3. Ethnicity of participant4. Age of infant5. Views about infants considered 'high risk' at birth i.e. those born too small or too early, or who were placed in a neonatal intensive care unit (NICU)6. Weight/growth status of infant after birth7. Mode of infant feeding Using agreed versions of quality appraisal and extracted data a textual narrative synthesis was undertaken by a single researcher (PL). Each study within a sub-group was described in a commentary reporting on study characteristics, context, quality, and findings. The scope, differences and similarities among studies were used to draw conclusions across the studies (the synthesis). Drawing conclusions across studies was not always possible due to study heterogeneity and lack of data. A worked example of the process is shown in Table We noted that unrelated members of the public tended to prefer infants of mid-range body sizes, but the evidence to support this observation was thin. Families of children with poor growth were acutely aware of growth as a problem; they monitored growth and discussed it with others. They desired "normal" growth in their child, and looked for ways that they could interpret the infant's growth as normal (for example finding members of the extended family who were of similar body shape). The most common method of assessing size in all sub-groups was by comparison with others, although the use of growth charts and physical measurement were also important for those with children with poor growth including babies born too small or too early. However, growth and size in themselves were low among concerns about such 'high risk' babies. The predominance of those with 'high risk' infants may explain our conclusion that growth was low among priorities for mothers of younger infants (aged 0–3 and 3–6 months). Among older children (more than 12 months) with poor growth there was concern among parents. Parents wanted to see good growth in their children, but they also considered love, attention, good health and good diet as important.We judged that we had insufficient data to draw conclusions about the views of family members other than mothers, health professionals, or to compare the views of participants of different weight, ethnicity, or toward breast versus bottle fed infants.Thematic synthesis was undertaken by two researchers, LA and PL. Findings from all studies were collated under the 7 questions used in data extraction. Each researcher independently conducted a thematic analysis using these findings. On initial discussion of themes, researchers judged that there was repetition between the data extraction questions, and that data referred to four broad areas of enquiry:1. Understanding healthy growth/size2. Assessment of growth/size3. Concerns about growth/size4. Influences on views, behaviour, interpretations of growth/sizeData and themes were grouped into these areas and emerging themes were then considered for relevance, presence across studies, 'thickness' and duplication. This process was repeated until researchers were satisfied that all data could be interpreted within these themes and an agreed version reached. A worked example of the process is shown in Table Across the thematic synthesis the predominant concern of participants was normality. This was seen through the creation of norms of growth and models to explain difference. This was conducted across physical, observable characteristics, but included physical unobservable  and non physical  dimensions. Where growth differed from the norm and a plausible explanation could not be found, for example among families of those with faltering growth , growth Data from across studies could be usefully combined in this method, for example in listing all the sources of influence on behaviour or views found. Family, other parents and friends, information from the infant themselves, health professionals, clothing sizes, magazines, books, radio, TV and their religious beliefs were all important to some, but the relative importance of these could not be explored.While the data extraction and thematic synthesis was undertaken by two researchers working independently, only one of these researchers  worked on the narrative synthesis with a second researcher discussing the work as it progressed. Whether the findings might be different with more than one researcher working on both syntheses, or researchers not involved in the data extraction doing the syntheses, or the syntheses being carried out in a different order, are themselves research-able (if rather expensive) questions, as is the issue of whether the immersion of one researcher in the data at every stage a strength (as we believe it to be) or a source of bias.Reassuringly, the conclusions to which these analyses led us about lay perspectives were largely similar across the thematic and textual narrative synthesis. Whether using a different research team, or a larger number of reviewers, would have produced different results is itself a researchable question. However, in this case conclusions from both analyses were dominated by importance of having babies that were a 'normal' size, leading to interest in monitoring of growth in a number of ways and, sometimes, to concern that there was an underlying problem leading to 'abnormal' growth. While the general conclusions were the same, the process and the implications of the two types of synthesis differed.A textual narrative approach typically groups studies into more homogenous groups. This technique has been particularly successful in synthesising different types of research evidence . Examples include a number of reviews carried out by the Evidence for Policy and Practice Information and Co-ordinating Centre (EPPI-Centre) -23, reviIn our review, the textual synthesis proved a useful way to describe difference in the included studies, making explicit the diversity in study designs and contexts. The textual narrative review also described gaps in the literature, both by showing where evidence was absent and by making an evaluation of the strength of evidence in different areas. Using this method enabled us to comment on, for example, the ethnic uniformity of participants, and the lack of evidence collected regarding mode of feeding.However, transparency remained a problem. For example, decisions about which sub-groups to use for synthesis of individual studies rely on judgements, albeit ones which can be informed by the scientific literature and by lay views. While we sought to make the decision making process clear, interpretation and judgement, which are not fully susceptible to external scrutiny, lie at the heart of the process.The strengths of the thematic synthesis lie in its potential to draw conclusions based on common elements across otherwise heterogeneous studies. This synthesis is potentially more accessible for the reader than a textual synthesis. Conclusions from this thematic synthesis fulfil an important research aim of qualitative research in generating hypotheses, an area to which traditional systematic reviews are poorly suited .However, pooling findings in the thematic synthesis risks masking the shortcomings of the individual studies that make up the review. Although descriptions of study characteristics and quality appraisal were presented alongside synthesised findings, the synthesis process obscured these in the conclusions. We believe that further debate about the reliability of this approach would be useful. On the one hand, the hypotheses that emerge from this synthesis draw on a broader body of views than any single study  and may therefore increase reliability; on the other, we risk making strong conclusions based on a group of studies none of which is in itself reliable on the grounds of quality or diversity of context. This method may also be poor at examining contradictions, as well as commonalities, in the data and at highlighting gaps in the evidence.The selection of synthesis method for systematic reviews such as this may depend on the aims of the synthesis. For the purpose of generating future research hypotheses, the thematic synthesis appears to hold the greatest potential; describing common themes and providing a possible structure for new research. In contrast, the textual narrative synthesis might be better suited to reviews which aim to describe the existing body of literature; identifying the scope of what has been studied, the strength of evidence available, and gaps that need to be filled.The authors declare no competing interests.CL, JB, HR, obtained funding for the study. All authors were responsible for the concept and design of the study. PL, and HR carried out the review work with assistance from all other authors. PL, LA & HR were responsible for the interpretation of findings. PL and HR produced the first and subsequent drafts of the paper, all authors were responsible for critical revision of the manuscript.The pre-publication history for this paper can be accessed here:


Journal of Health Care for the Poor and Underserved aimed to determine whether the paucity of minorities included in research could be explained by differences in willingness and misgivings related to participation in health research.Medical research studies often do not include ethnic and racial minorities as study participants in numbers that are representative of their populations. A study published in the November 2006 issue of the The study, funded by the National Institute of Dental and Craniofacial Surgery, was conducted by a research team within the Tuskegee Legacy Project (TLP), which was inspired by a 1994 bioethics conference at the University of Virginia. The research team was created to assess how the infamous Tuskegee Syphilis Study affected the attitudes of black Americans toward health research. From 1932 to 1972, 399 black men with syphilis were studied to observe the effects of untreated syphilis, even though effective treatment was already available. This unethical study has often been used to explain the assumption that blacks may be more prone than whites to distrust research and refuse to participate.To test this assumption, the research team developed the TLP Questionnaire, which contained two scales: the Likelihood of Participation Scale and the Guinea Pig Fear Factor Scale, which measured self-reported general willingness to participate in research and fear of participating in research, respectively. The TLP Questionnaire was taken by more than 1,000 black, white, and Hispanic residents of four U.S. cities .Significantly, the results showed that only about 30% of all people surveyed expressed a willingness to participate in research studies. Blacks were 1.8 times as likely as whites to fear participating in biomedical research. Still, they were equally as willing to participate in research as whites. “Given the history of the treatment of African-Americans in our country, it makes sense that blacks would have a heightened awareness of potential dangers,” says lead author Ralph V. Katz, chairman of the Department of Epidemiology and Health Promotion at the New York University College of Dentistry.These findings are consistent with the few other studies in which racial/ethnic differences in research participation have been assessed. They also are particularly important in studies addressing health disparities and those that aim to study environmental justice issues in minority populations. “African Americans come from varied experiences in the health care system. As such, there is no monolithic response to health-seeking behaviors, including participation in health research,” says Ruth Browne, CEO of the Arthur Ashe Institute for Urban Health in Brooklyn, New York. “This really points to the importance of culturally appropriate outreach efforts.”


Generation Scotland: the Scottish Family Health Study aims to identify genetic variants accounting for variation in levels of quantitative traits underlying the major common complex diseases  in Scotland.Generation Scotland will recruit a family-based cohort of up to 50,000 individuals (comprising siblings and parent-offspring groups) across Scotland. It will be a six-year programme, beginning in Glasgow and Tayside in the first two years (Phase 1) before extending to other parts of Scotland in the remaining four years (Phase 2). In Phase 1, individuals aged between 35 and 55 years, living in the East and West of Scotland will be invited to participate, along with at least one (and preferably more) siblings and any other first degree relatives aged 18 or over. The total initial sample size will be 15,000 and it is planned that this will increase to 50,000 in Phase 2. All participants will be asked to contribute blood samples from which DNA will be extracted and stored for future investigation. The information from the DNA, along with answers to a life-style and medical history questionnaire, clinical and biochemical measurements taken at the time of donation, and subsequent health developments over the life course  will be stored and used for research purposes. In addition, a detailed public consultation process will begin that will allow respondents' views to shape and develop the study. This is an important aspect to the research, and forms the continuation of a long-term parallel engagement process.As well as gene identification, the family-based study design will allow measurement of the heritability and familial aggregation of relevant quantitative traits, and the study of how genetic effects may vary by parent-of-origin. Long-term potential outcomes of this research include the targeting of disease prevention and treatment, and the development of screening tools based on the new genetic information. This study approach is complementary to other population-based genetic epidemiology studies, such as UK Biobank, which are established primarily to characterise genes and genetic risk in the population. Most medical disorders of public health importance have a significant heritable component. This includes cancer, heart disease/stroke and mental health  , as wellGeneration Scotland (GS) is a collaborative initiative between the Universities of Aberdeen, Dundee, Edinburgh, Glasgow and St Andrews, and the Medical Research Council Human Genetics Unit, the National eScience Centre, the Scottish School of Primary Care, and the National Health Service (NHS) Scotland. It is funded by the Scottish Funding Council (formerly Scottish Higher Education Funding Council), and aims to promote research into genetics and healthcare . GeneratThe aim of GS:SFHS is to establish a new, large, family-based intensively-phenotyped cohort recruited from the general population across Scotland, as a resource for studying the genetics of health areas of current and projected public health importance. DNA and non-identifiable information from this cohort will be made available to researchers in Scotland and international collaborators. It will be a six-year fieldwork programme, beginning in Glasgow and Tayside in the first two years (Phase 1) before extending to other parts of Scotland in the remaining four years (Phase 2).The objectives are:Recruitment. To recruit and phenotype a family-based cohort from across Scotland, allowing identification of genetic variants relevant to the pathogenesis of common complex diseases  and pharmacogenetics research.1. Public Understanding. To conduct an early and sustained public consultation programme to understand and explain the public reaction to genetics in healthcare, and their reaction to participation in research.2. Research capacity. To create a multi-institutional and NHS collaboration across Scotland that will share knowledge and best practice in human genetics research.3. Health informatics. Through this collaboration, to create a nationwide research platform in emerging technologies of health informatics in genetics research, including the linkage of study data to "real-time" routine healthcare data on an individual basis.4. NHS integration. To work with NHS Clinical Genetics colleagues to develop computer-delivered education on genetics for healthcare professionals in Scotland.5. Exemplar studies. To conduct specific research projects with identified protocols in the areas of cardiovascular disease, mental health, osteoporosis and pharmacogenetics, using the resource that will be established.6. This paper describes the methodology for the first of these objectives, currently in its pilot phase, though it is important that all of the objectives are closely integrated. In particular, findings from the public consultation will contribute directly to continuous development of the materials and methods used to recruit the cohort, and the study will undergo periodic review in light of the findings of this parallel component. This will include study of potential participants' views on recruitment, consent, withdrawal, feedback, confidentiality, storage of and access to bioinformation, commercialisation, governance and other associated ethical, legal and social issues. This article therefore discusses the structure and form of a nascent GS.This is a family-based study, recruiting participants and their relatives mainly through primary care. Recruitment of general practices is facilitated by Scottish Practices and Professionals Involved in Research (SPPIRe) . PotentiAn independent party, based in the NHS, generates a list of eligible people registered with each collaborating general practice, from the Scottish NHS register ). The names of all potential participants are screened by their GP, and individuals whom it might be inappropriate to approach  are excluded. Letters of invitation to eligible participants are generated on practice headed note-paper and signed by one of its GP Principals. These letters are dispatched by the independent party by post, with up to two reminders as required. This invitation is for agreement to discuss the study with family members with a view to possible participation. When the eligible person returns the tear-off slip agreeing to be contacted by the research team the individual's details are sent from the independent party to the study team.In addition, targeted approaches in the Tayside area will be made to potentially eligible individuals whose details are held on the Walker Birth Cohort database . This isUpon receiving their permission, potential participants are contacted by a member of the research team to ensure that participants understand the study, that all demographics and details are accurate, and to discuss participation in the study with the relevant first degree relatives (including at least one sibling group). The names and contact details are requested of all first degree relatives who have verbally indicated, to the individual initially contacted, their willingness to be approached by the research team. These relatives are then also contacted by telephone by the study team. Each relative contacted is invited to discuss with and identify further first degree relatives, and so on, with the aim of creating a "snowball" sampling effect.et al). Although it is the main method of approach and recruitment, it will be augmented by a programme of communication and publicity about the study. Throughout this programme, individual families will be invited to volunteer directly for the study, by contacting the research team. They will be able to participate if the family includes at least one sibling pair.Similar methods of approaching and recruiting participants and their relatives have been successfully used in other studies in which several of the co-applicants have been closely involved. These include the British Genetics of Hypertension (BRIGHT) study  and "FamFollowing verbal consent, individuals and their relatives who agree to consider participation in the clinical study are sent a copy of the Participant Information Leaflet (PIL), a consent form, and a pre-clinic questionnaire (PCQ), and arrange for attendance at a research clinic at a mutually convenient time and location. They are asked to bring these and any medications taken regularly, to the clinic visit. If possible, this appointment is at a time that allows collection of a fasting blood sample. The feasibility of this is being reviewed as the study progresses.On attendance at the clinic the consent form is discussed and completed prior to any sample or medical details being taken. The participant then hands over the completed PCQ, with the opportunity for clarification of any items with the research nurse, has clinical and interview measurements made, and provides blood and urine samples, all according to detailed Standard Operating Procedures (SOPs). This visit lasts between 90 minutes and two hours.The data collected aim to maximise the research benefit of each individual's attendance while retaining efficiency and acceptability. As well as the PCQ, they include biometric measurements, tests of cognitive function, questionnaires relating to personality traits and psychological distress, and screening for major mental illness Table . ValidatDemographic, clinical and biological data are entered initially on a paper clinical reporting form (CRF), but kept separately from any personal details that would allow identification of individuals, using a barcode system. The PCQ and CRF are scanned into an electronic database, with detailed in-built validity checks of data recording. Feedback is given to participants and, with their permission, their GP, in the form of basic clinical information . ECGs are reported centrally, and summarised results are also fed back to GPs, with permission. Detailed results of these are returned to the research team for entry to the database, and summarised results are fed back to GPs (with the participants' consent), along with basic clinical recommendations based on these.The primary objective of this study is to establish the cohort and database as a resource for future analysis. It is important to maximise the utility of the resource, and some general principles of analysis have been determined. We propose to adopt both variance components linkage and regression approaches to the genetic linkage analysis of continuous quantitative trait data (QT) since these have been shown to be powerful approaches . This wi15,000   sibpairs, thus requiring recruitment of 30,000   individuals4,700   sibtrios thus requiring recruitment of 14,400   individuals2,400  [625] sibquads thus requiring recruitment of 9,600   individuals78 (38)  pedigreeOur recruitment is based on families with a minimum of a sibpair. However, we seek to recruit all siblings within families that are recruited, and aim to recruit as many sibtrios as possible. Recruitment of parents and adult offspring will further increase study power and will permit future family-based association studies and haplotype analysis. We therefore also seek to recruit one or both parents, wherever possible. If we take the power estimates for sibtrios as our main estimate then in ideal circumstances we will require to recruit 14,400  sibs to be able to identify a QTL with an effect corresponding to QTL heritability of 10% (15%). The proposed cohort size over the first 2 years will effectively be between these two estimates. Actual study power will be increased by the inclusion of parental data and will increase in proportion to our success in recruiting some larger pedigrees but will be decreased by any genotyping errors , QT measurement error and imperfect reliability (repeatability) of QT measures. We believe that a study power calculation based on that for sibtrios is a reasonable compromise and suggest that the study will have 80% power to detect a QTL heritability of between 10% and 15%. We will further maximise study power by selecting traits for study based on their known (high) heritability and repeatability. If we are successful in targeting and recruiting larger families then the above power calculation will be very conservative and significantly underestimate the true study power. As recruitment rises towards 50,000 in years three to six this will give study power to detect QTL heritability of between 5% and 10%.Questionnaire and interview data are optically scanned in to a secure database with a unique study number identifying each individual. Biochemical samples are processed by local NHS laboratories, and the data entered on to the same secure database. Further biochemical samples (serum and urine) are processed locally in research labs and archived for future analysis. A 9 ml EDTA blood sample will be delivered (in batches of frozen tubes) to the Wellcome Trust Clinical Research Facility in Edinburgh , for DNAAn important part of GS:SFHS is the development of an IT infra-structure that can support the research, from identification of potential participants to linkage of data with other useful databases. This both facilitates the study and maximises its value. A suite of Windows-based applications and services, implemented with web services, provides access to IT facilities for identification, approach and recruitment of subjects, appointments, consent, storage of paper-based data, and feedback of clinical information. Participants are identified by a unique barcode, with systems to ensure efficient and confidential handling of all data.Through the CHI numbers of participants, and by a process of encryption, study data will be linked to a number of routine datasets, including virtually all formal healthcare activity Table . ConsentAll components of GS:SFHS, including the protocol and written study materials, have received formal, national ethical approval from NHS Tayside Research Ethics Committee (REC reference number 05/S1401/89). In addition local approval has been obtained from NHS Glasgow Research Ethics Committee, and from NHS Glasgow and NHS Tayside Research and Development Offices, as is required.The study aims to minimise several risks to participants.1. Direct researcher contact maximises subject accrual but at the risk of compromising privacy and confidentiality. The use of an independent party to work between the research team and participating general practices is designed to minimise the risk to the privacy of potential participants. We need to ascertain what the actual recruitment rate will be, and the incorporation of the public consultation study will ascertain what issues are likely to affect participation, and how these might be addressed.2. Heightened risk of disclosing confidential information. No information is given by the research team to probands or relatives about each other, and only basic lifestyle and health information will be fed back to participants. Separate discussions between the research nurse and both the proband and the relative(s) maintain confidentiality, and allow individual concerns to be addressed and a private and frank discussion about participation to ensue.3. Because it is impossible to know what precise purposes the resources will be used for in the future, fully informed consent for the use of data and DNA cannot be obtained. Because of the need to maintain participant anonymity and confidentiality, the logistical difficulties of re-tracing participants and the annoyance factor of persistent re-consenting, currently "blanket" consent is sought, for full research and commercialisation purposes.1. Despite this being a family study, individual confidentiality remains paramount and no medical information is given to individuals or relatives about themselves or each other, apart from personal lifestyle and basic clinical information to individuals who request this at the time of collection.2. We emphasise that participation is at all times voluntary. Withdrawal will be allowed at any time, upon which all data and samples, that have not been analysed, relating to the withdrawing participant will be destroyed.3. Bias will be introduced into both the clinical and consultation study in recruitment of family members by the proband. Family is not purely about biological connections but social and kinship networks. The design offers no option other than to go with the family's choice in who can participate, and their stated biological relationships with each other.4. No genetic feedback will be given to participants; this is a project that aims to construct a database for research purposes and does not purport to offer genetic tests either to individuals or populations.5. Access to the samples and data by third parties will be reviewed by GS:SFHS senior management groups, subject to an independent governance system whose details are to be finalised in light of the public consultation study and external stakeholder advice, and NHS Research Ethics Committees. Data will only be made available to outside parties after personal identifying information has been removed and/or securely encrypted.The REC will be informed of progress and proposed modifications to the protocol. In addition, an independent GS Advisory Board has been established under the auspices of SEHD CSO, and will monitor the study's procedures, protocols and data storage and access. This will be analogous to a Data Monitoring Committee in a clinical trial, and will have right of access to all study materials (anonymised when appropriate).Unlike general population biobank studies, such as UK Biobank , GS:SFHSThe Scottish population (currently 5.1 million approximately ) is ideaGS:SFHS is one of a growing number of large DNA collections or biobanks across the world. Two important unique characteristics of GS:SFHS will be (i) the large general population family-based nature, and (ii) the ability to link individual data with detailed past and future health records. It is also unique for its inclusion of detailed cognitive function phenotype information, and its collection of data on serious mental illness, both of which will provide rich opportunities to study the genetics of these health areas.The key parts of GS:SFHS are the desire to intensively phenotype family members at baseline and to follow up participants through routine data systems and to seek their consent for further fieldwork. Scotland is uniquely well-placed to allow linkage of participants' collected data with routine data from the NHS because of the widespread use of unique CHI numbers linking individuals, through the CHI, with many other NHS datasets. A bioinformatic infrastructure is well-developed to support research that maximizes the value of this while retaining the anonymity of participants. Tayside is currently the only region in the UK where the CHI number is used as the patient identifier in the virtually all health care activities, from primary to tertiary care, from birth to death, and is added routinely to all research datasets in order to enable further phenotype enrichment. A core aim of the SFHS is to export expertise in Tayside to other centres in Scotland.Linkage analysis has proven to be very effective for identifying genes concerned with rare Mendelian disorders and Mendelian forms of genetically complex ones. Thus, it is the major strategy used for identification of genetic variants of large effect.However large genetic effects tend to result in selection against these alleles, therefore causing only rare diseases or rare forms of common diseases. The low power to detect genetic variants of small effect has suggested that this approach may be much less effective for common complex diseases. However, recent pedigree-based studies carried out by deCODE Genetics in Iceland have reported promising results with denser marker scans and larger pedigrees than in past studies . GS:SFHSmoderate rather than large  or small (unlikely to be detectable by this approach) effect sizes. It is likely that genetic factors with large and moderately large effects on complex traits or disease risk code for proteins involved in important physiological mechanisms. In addition therefore, they are likely to be important both in terms of our understanding of pathological mechanisms and of their potential to lead to new prevention or treatment strategies.The focus of the study is to identify genetic variants of This pedigree or family-based approach has a number of other advantages:familial correlations to increase power using strategies such as the EDAC ("extreme discordant and concordant") design.• the ability to make use of information on genetic haplotypes .• the ability to obtain transmission-disequilibrium test (TDT) by comparing transmitted with non-transmitted parental alleles.• the ability to carry out a family based association approach including using the parent-of-origin information .• the ability to provide Most family-based collections have been affected sibpair studies , for a nIn summary, the family-based design of this study will permit both linkage and family-based association study approaches and the large sample size will give power to detect moderate genetic effects on QT levels ,26. The GS:SFHS is actively recruiting to the Phase 1 in both Tayside and Glasgow. It is intended that data from these participants will contribute to the main study database. A review of the Phase 1 will provide information on, among other factors, recruitment and participation rates, pedigree size and nature , and planning for Phase 2 of the study across the whole of Scotland. These data will be written up for publication as the study progresses .SHR holds patents on genetic markers for osteoporosis susceptibility. There are no competing interests for any other author.All authors contributed to the writing of the study protocol, in an iterative manner. The main text was written by BHS and HC, with comments and amendments made by all authors, who have each read and approved the current version of the paper.The pre-publication history for this paper can be accessed here:


The increase in childhood obesity and prevalence of chronic disease risk factors demonstrate the importance of creating healthy school environments. As part of the Border Health Strategic Initiative, the School Health Index was implemented in public schools in two counties along the Arizona, United States-Sonora, Mexico border. Developed in 2000 by the Centers for Disease Control and Prevention, the School Health Index offers a guide to assist schools in evaluating and improving opportunities for physical activity and good nutrition for their students.Between 2000 and 2003, a total of 13 schools from five school districts in two counties participated in the School Health Index project despite academic pressures and limited resources.The Border Health Strategic Initiative supported the hiring and training of an external coordinator in each county who was not part of the school system but who was an employee in an established community-based organization. The coordinators worked with the schools to implement the School Health Index, to develop action plans, and to monitor progress toward these goals.The School Health Index process and school team participation varied from school to school. Individual plans were different but all focused on reducing in-school access to unhealthy foods, identified as high-fat and/or of low nutritional value. Ideas for acting on this focus ranged from changing the content of school lunches to discontinuing the use of nonnutritious foods as classroom rewards. All plans included recommendations that could be implemented immediately as well as those that would require planning and perhaps the formation and assistance of a subcommittee .After working with the School Health Index, most schools made at least one immediate change in their school environments. The external coordinator was essential to keeping the School Health Index results and action plans on the agendas of school administrators, especially during periods of staff turnover. Staff turnover, lack of time, and limited resources resulted in few schools achieving longer-term policy changes. Adult U.S. Hispanic populations living along the Arizona, United States-Sonora, Mexico border experience type 2 diabetes prevalence rates that are double the rate of the general U.S. population ,2. The rIncreases in rates of diabetes are closely associated with obesity rates. Obesity rates among U.S. children have been escalating rapidly over the past three decades ,6,7. DatSchools are ideal environments for promoting physical activity and good nutrition ,10. UnfoTo address physical activity and nutrition in the school environment, the CDC developed the School Health Index for Physical Activity and Healthy Eating: A Self-Assessment and Planning Guide (SHI) in 2000 To addresBorder Health ¡SI!)  . The majBorder Health ¡SI! recruited eight schools from three public school districts in the Nogales area. Combined, these districts serve approximately 9256 students and have 10 elementary schools, three middle schools, and three high schools. Of these 16 schools, six did not meet the federal Leave No Child Behind criteria in 2003, and two were underperforming  kept the year-round schedule.rforming -17. No sIn the Yuma area, five schools were recruited from the communities of Somerton and San Luis, which are 100% Hispanic. Combined, the two communities had a population of approximately 24,610 in 2001 ,19. BetwBorder Health ¡SI! worked with two public school districts in Yuma County. At that time, the districts served approximately 6524 students from K–12, with six elementary schools, two middle schools, and one high school. Of the nine schools, four were ranked by the state of Arizona as underperforming, and four did not meet the federal Leave No Child Behind criteria in 2003 , selected the recently released SHI as a tool that would enable schools to start thinking about creating healthier environments. Despite the lack of published evaluation results, the Border Health ¡SI! group felt that it was a reasonable tool to focus schools on physical activity and nutrition policy. The SHI and follow-up were the only school-based interventions as part of Border Health ¡SI!.The community-based agencies involved with The University of Arizona Cooperative Extension in Yuma County and Southeast Arizona Area Health Education Center (SEAHEC) in Santa Cruz County facilitated implementation of the SHI. These two community-based agencies had strong existing relationships with local schools. SEAHEC was involved in a variety of school nutrition education programs, and Cooperative Extension was responsible for the 4-H clubs for children and thus also worked closely with area schools.Border Health ¡SI! expressed concern that the schools were overburdened and that health promotion and chronic disease prevention might not be high priorities. We believed, however, that an outside advocate could discuss the serious issues related to chronic disease and how they impact children. We also believed that resource-stressed schools would accept external coordinators from established and trusted agencies to provide assistance and support. Cooperative Extension and SEAHEC identified staff members who could serve as external coordinators to assist schools in completing the SHI assessment and planning process and in coordinating and compiling the SHI materials. MEZACOPH staff provided education to external coordinators on the SHI and the relationship between adolescent health and chronic disease. External coordinators were responsible for documenting recruitment efforts, the SHI process within the schools, team member activities, and action plans.The community agencies and others involved in External coordinators initiated the recruitment process by presenting the SHI to school district superintendents or assistant superintendents, the school boards, or directly to principals. In addition, external coordinators contacted schools where they had personal connections. By the end of the third year, all schools in the area were approached, and any that expressed interest were contacted. The external coordinator provided a verbal overview and copy of the SHI, and if received positively, made a presentation to school personnel. When schools were hesitant to participate, additional assistance was sought from two directors of health services, a school board president, and a registered nurse at a school-based clinic to encourage schools to participate. Schools were offered a financial incentive of $1500 upon completion of the SHI. The incentive was provided by Cooperative Extension and SEAHEC. Schools were encouraged to apply the funds toward their action plans but were not required to do so. At the end of the three-year period, the SHI was implemented in 10 elementary schools, two middle schools, and one high school, about half of the 25 schools approached.Once a school agreed to complete the SHI, the principals identified an internal SHI coordinator. The internal coordinator recruited team members, and the external coordinator met with the team to provide materials and an overview and outline the assessment process. The external coordinator then followed up with the internal coordinator to ensure that the teams were meeting, collected scorecards, produced a summary document, and scheduled and helped conduct the action planning session.Border Health ¡SI! was a legislative appropriation and was not funded as a research project, resources were not available to do an in-depth study of the SHI. However, the community-based agencies and MEZACOPH reached an agreement on an evaluation plan. Documentation of the plan would include information on the interest schools showed in completing the SHI, the ability of SHI teams to understand and complete the SHI, the process for completing the evaluation instrument, whether or not the teams created action plans, and finally, whether or not schools were able to make any changes suggested in the action plans.Because Detailed quarterly reports by Cooperative Extension and SEAHEC were used to document school interest and the SHI process. MEZACOPH staff conducted in-depth interviews with the external coordinators and with a convenience sample of 14 SHI team members from the first five schools to complete the SHI. The purpose of the interview was to identify barriers to implementing the SHI, to find out whether or not SHI team members believed an external coordinator was necessary, and to identify changes in the school environment that could be attributed to the process. The external coordinators were in frequent contact with the schools after completion of the SHI. A year after completion of the SHI, external coordinators contacted the schools to determine whether or not SHI action plans were being implemented.The SHI process was different in each of the schools. The time frame for complete implementation ranged from four weeks to almost two academic years. Most schools completed the SHI within one semester. Schools were encouraged to complete the SHI during the fall semester to allow for time to work on action plans. Unfortunately, all schools completed the SHI during the spring semester.The SHI team composition varied by school. Team size ranged from six to 34. All schools included at least one parent. As described earlier, the border communities are predominantly Hispanic; therefore, at several schools, parents emphasized the need for a Spanish version of the SHI. A health educator and social worker were not included for Yuma area schools because these positions do not exist in these schools.An in-depth interview with 14 SHI team members revealed that team members felt that the SHI helped to build awareness of school commitment, identify changes that do not require resources, encourage policy and action, bring health issues to the schools' attention, and raise awareness of federal policies. The team members also identified the four key barriers to implementing the SHI: 1) time, 2) getting people to meetings, 3) initial buy-in, and 4) perceived lack of expertise. The SHI team members believed that the key roles played by the external coordinators were facilitation and guidance. The external coordinators also assisted in overcoming barriers.Border Health ¡SI!, community coalitions or Special Action Groups (SAGs) were established to focus on policy change to create healthier communities  used SHI results to develop the district's comprehensive health plan. Results were not available from the individual schools, and the district health plan is not complete at this writing. The information reported here is based on reports from three of the elementary schools representing two school districts. Overall, half of the schools in the area completed the SHI. The main reasons for schools refusing to participate were lack of time and lack of a school champion. When a school champion was identified, time became less of a barrier.By the end of The intent of the SHI is for SHI teams to collaboratively develop action plans based on the results from the eight SHI modules. In two of the Santa Cruz County schools, the SHI was completed at the end of the semester, and the teams were unable to develop action plans. The principals, who had participated on the teams, created the action plans in isolation. This process was especially problematic because one of the principals resigned shortly after creating a plan. In fact, by the beginning of the next academic year, only two of the seven SHI team members were still at their school (hereafter referred to as Santa Cruz School 1 [SC1]). The second principal to develop action plans promoted many school-level changes (Santa Cruz School 2 [SC2]). Unfortunately, she also left her position.salditos  instead of baked goods and junk food.SC1  made several changes as it implemented the SHI, including the decision to move all candy sales out of the cafeteria. Most notably, the school hired a full-time PE teacher and developed a policy that prohibited the selling of junk food for fundraisers. The fundraising policy came into question the following academic year when the loss of almost all members of the SHI resulted in very little institutional memory. This case offers an example of how important the external coordinator's role is in the process. The remaining SHI team member was uncomfortable promoting the action plans to the new principal. The external coordinator contacted the principal early in the school year to discuss the SHI and action plans. The long-term goal of the SHI team was to increase the length of the lunch period. The principal was unfamiliar with the SHI but expressed verbal support for the action plans. When resistance developed over the fundraising policy, the principal enlisted the aid of a remaining SHI team member to explain the policy to the staff and parent-teacher organization. The policy was upheld. SC1 had a new principal the following year. The external coordinator continued to follow up and provided the impetus for the principal to create a new SHI team that completed the SHI a second time. SC1 has had a full-time PE teacher for three years. They have not lengthened the school lunch period. Fundraisers now include gifts, wrapping paper, and magazines as well as chocolate. Items for sale at lunch include pencils, notebooks, pickles, oranges, peanut butter crackers, and Border Health ¡SI! coalition for assistance. Coalition members, teachers, and parents attended the school board meeting. The principal presented the SHI results, and the teachers and parents strongly supported the program. The PE program was not cut. Unfortunately, before the next academic year, the principal accepted a job as a district superintendent in another community. In that role, she was able to add PE into all elementary schools. The PE course is pass/fail. Individual teachers are offering low-fat snacks and less sweet snacks at parties, but this is not a coordinated school effort. Carrots, orange juice, milk, graham crackers, and cheese sandwiches are served at the school open house.The principal/superintendent at SC2 was deeply committed to the SHI process. The school made several immediate changes. The school bake sales were converted to healthy snack sales. Graduation cookies and punch were cancelled and replaced with lemonade and baked chips and salsa. As a result of the SHI, the school hired a full-time PE teacher and developed a PE course. The goal was to have a letter grade for the course, not a pass/fail grade. An existing staff member was certified as a PE instructor. The school also organized track and basketball teams for the first time. The following summer, the school board attempted to drop the program. The principal called the Border Health ¡SI! coalition. The team was encouraged by the coalition to write a newspaper article for the local paper describing the changes they were making. Students were interviewed by a local television station when the soda was replaced by fruit drinks. The students said they missed the soda "but juice was okay."The third Santa Cruz school (SC3) to complete the SHI took one and a half years to complete the process. The school was undergoing major renovations when it was first contacted and despite expressing interest, it was unable to commit to the process. Once the school committed, however, the external coordinator reported that the SC3 SHI team was the most enthusiastic. The school removed the vending machine from the cafeteria and decided to remove all other soda and candy vending machines and replace them with healthier choices. The beverage machines include fruit juices, water, tea, and lemonade. The SHI team also recommended removing the school store from the cafeteria. The SHI team reported their results to the The school store has not moved, but it is no longer selling candy. It replaced candy with healthier options, and profits have stayed the same. Bake sales now have oranges, cucumbers, and carrots. Some sales include candy but only small candy bars.Border Health ¡SI! in the southern Yuma area. All three elementary schools in one district participated. In the second district, only one elementary school participated. It was the only elementary school not running double shifts. Overall, five of the nine schools in the area completed the SHI. The main reason for refusing to participate was lack of time. In Yuma, the external coordinators actively facilitated the action planning process. Documentation from the Yuma area focuses on the action plans.A total of five schools, four elementary schools and one middle school, implemented the SHI as part of the All teachers participated on the SHI teams at the first school (YC1) in the Yuma area. Not all teachers were enthusiastic. A school policy was developed to prohibit the use of food as a reward in the classroom, and students were limited to one snack per day. The SHI team also set goals of 1) teaching more nutrition information, 2) gathering and disseminating information on all food and drink items sold at the school, and 3) shifting from selling junk food items and sports drinks to healthier options. The school offered granola and fruit bars as alternatives, but they did not sell, and the school reverted to selling junk food. YC1 established walking groups two to three days per week for students. Teachers organized structured fitness breaks. The school also implemented an annual field day for staff and students.YC2 also established a school policy prohibiting nonnutritious food as a reward and limiting snacks to one per day per student. The SHI team was especially interested in eliminating outdated health education material and incorporating a sequential health education curriculum. Teachers began standing at the salad bar to encourage children to eat more fruit and vegetables. The school also began to increase use of community facilities and to offer a swimming class at the local community pool. The school's part-time PE teacher left the school for a full-time position out of the district. The SHI team presented the SHI results at a staff workshop.YC3 established a lengthy list of goals, including requiring hand washing, adopting a sequential health education curriculum, and incorporating activity breaks into the classroom. Two years after implementation of the SHI, no progress has been made.Border Health ¡SI! coalition.The three Yuma area elementary schools described here are from one school district. The superintendent and SHI team members requested that the external coordinators from Cooperative Extension present the SHI results to the school board. The perception was that the board would view the external coordinators as community members and not as school personnel, and they would thus have a greater impact. The SHI team members then presented the results to the The fourth Yuma area elementary school (YC4) was from the second school district and focused on similar issues as the other three schools. This school chose to focus on increasing instruction time for health and PE, adopting new health education textbooks, and presenting health-related information to parents and staff. At this point, no progress has been made toward these goals.The action plans of these schools emphasized modifying health-related curricula and adopting a sequential health education curriculum. Upon exploring the issue at the district level, it was determined that the district is not considering modifications to health education curriculum until 2005. To provide the teachers with resources, Cooperative Extension presented a resource event where teachers selected educational materials focusing on health education, nutrition, and physical activity and provided suggestions on ways to incorporate nutrition messages and physical activity into the existing curricula. In addition, Cooperative Extension also provided one school library with more than 50 books on physical activity and nutrition (fiction and nonfiction).Seven Arizona elementary schools along the Arizona-Sonora border assessed their school environments and developed action plans using the CDC's SHI. Despite some difficulties resulting from time constraints and human resources, all schools were able to complete the assessment. The schools focused their action plan priorities on nutrition and physical activity. School action plans included items that could be addressed immediately  as well as policies that would necessitate implementation at the district level . The individual school action plans varied, but all seven shared one component: to reduce internal access to unhealthy foods. Plans included changing the content of school lunches, discontinuing the use of nonnutritious foods as classroom rewards, moving candy sales and a snack bar away from the cafeteria, and choosing healthy alternative fundraisers. Two schools were able to use SHI results to hire PE teachers.Border Health ¡SI! was for schools to disseminate the results of the SHI at least to the teachers at each participating school. In addition, we hoped that the results would be presented at parent-teacher and school board meetings. High rates of staff turnover highlight the importance of disseminating SHI results and action plans to a larger audience. It is crucial for schools to have an advocate for physical activity and nutrition. The external coordinator and Border Health ¡SI! coalitions served as those advocates and were able to keep the issue of school health prominent in school administrators' minds.The goal of Border Health ¡SI! SAGs.The number of underperforming schools in these districts and the lack of highly performing or excelling schools results in stress on the educational system. Principals and school board members are under pressure to improve the academic performance of their schools. When approached for funds or time to implement new programs, school administrators frequently cite the need for scholastic improvement above all other issues. External coordinators returned repeatedly to meet with school administrators to discuss the importance of physical activity and good nutrition. An additional tactic for educating administrators was to recruit them to participate in the The current educational system is responsible for a wide variety of activities: academic performance, social services, childhood immunizations, and the nutrition of students through the federal school breakfast and lunch programs. Involving schools in health promotion and disease prevention activities is critical, but we must recognize that most school systems are under extreme pressure to demonstrate academic progress. The fact that half of schools approached in two low socioeconomic areas participated in the SHI process indicates the commitment of school personnel to the overall health and well-being of their students and communities. The support of an external coordinator from a local agency can assist in removing some logistical barriers.While most schools were extremely committed and enthusiastic about SHI action plans, staff turnover, time, and limited resources were barriers to progress even with the support of an external facilitator. Implementation of new programs is limited further by the low number of certified PE and health education specialists employed by the districts. Change in staff in one district occurred during the first year, and staff had to be educated about the SHI action plans and school goals. Some schools were undergoing major renovations and building projects, and in some cases, new schools were built. These schools found it challenging to take on new projects like the SHI.Border Health ¡SI! SAGs on how to convey our messages. The negative impressions receded after a brief time, and five more schools completed the SHI by the end of Border Health ¡SI!.One cautionary note is that publicity may present a barrier to acceptance of the SHI process. In the opinion of the external coordinators, publicity surrounding the removal of soda machines at one school may have discouraged other schools from participating in the SHI process. When funding for schools is so tight that vending machine sales are used to support photocopying expenses, field trips, graduation ceremonies, and extracurricular events, the threat of losing those funds can deter schools from eliminating this source of revenue, even though they are interested in improving the health of their students. The SHI and our project did not push schools to remove vending machines; instead, we encouraged schools to identify the best priorities for their own schools. These messages were lost by the news media. The external coordinator sought advice from the This project showed the value of having an external coordinator to help with continuity and with keeping the project top-of-mind with school officials, especially during periods of high staff turnover. In addition, the external coordinators acted as a resource beyond coordination. External coordinators created resource manuals for alternative ideas for school fundraisers, linked schools with other community resources, found funds to provide teachers with educational materials, and volunteered at school events. Although the SHI process does not include incentives, we found that monetary incentives for carrying out policy priorities seemed to encourage participation and gave schools resources to implement policies. Overall, most schools were able to implement immediate changes. Policies requiring a longer-term process and additional resources were more difficult to carry out. Future projects should focus on documenting whether students increased their physical activity or improved their eating habits as a result of SHI policies.


There is a large increase in back pain reporting in the early teens. In no previous study has the prevalence of low back pain been investigated in relation to the onset of puberty. The objective of this study was to establish whether the onset of puberty is associated with back pain reporting in young girls.A subsample of 254 girls aged 8–10 years and 165 girls aged 14–16 years from a cross-sectional survey of 481 children aged 8–10 years and 325 adolescents aged 14–16 years of both sexes.Main outcome measures were back pain defined as low back pain, mid back pain, and/or neck pain in the past month.Other variables of interest were Puberty (five different stages), age, body mass index, and smoking. Independent information on onset of puberty was obtained through a physical examination and on back pain through an individual structured interview. The association was studied between onset of puberty and the outcome variable , controlling for overweight, and smoking. Odds ratios with 95% confidence intervals were used to describe bivariate associations, logistic regression with robust standard errors was used for multivariate analyses.There is a highly significant trend for increased back pain reporting with increasing level of puberty until maturity is reached. The biggest leap appears between the second level (beginning of puberty) and the third level (mid puberty) and the findings remain after controlling for the covariates. These results emanate from the low back, whereas pain in the mid back and neck do not seem to be linked with pubertal stage.In girls, the reporting of low back pain increases in frequency during puberty until maturity, regardless of age. Why some girls are susceptible to back pain in the early stage of puberty is unknown. Back pain has been shown to commence early in life ,2. In a Attempts have been made to identify the age, when back pain becomes common and, at least in the case of pain for the lower back, there seems to be a rapid increase in the back pain reporting around the age of puberty . If the 3) and have been summarized in Appendix 1. A subsample of 254 female children and 165 female adolescents was selected from the participants in a randomly selected sample of Danish school-children attending elementary school (i.e. children aged 8–10 years and adolescents aged 14–16 years). These had been interviewed with help of a semi-structured questionnaire by one of the authors (NW) for the presence of back pain.The study design and methods have been extensively described elsewhere  reported to have occurred on the day of the study, in the week preceding the interview, or in the month preceding the interview, i.e. the one-month period prevalence of back pain.Start of puberty was assessed according to Tanner  based onData on back pain and puberty were collected by two independent examiners who were naïve in relation to the possible link between puberty and back pain, as the idea for the present project arose after the publication of a first set of articles, and the purpose of the main study was to determine various biological risk factors for cardiovascular disease.One at the time, the four different definitions of back pain  were cross-tabulated against the pubertal stage variable and reported as odds ratios with 95% confidence intervals.These associations were tested for trends across ordered groups with a score test for trend of odds . The scoTo take into account the possible effect of cluster sampling (independence of observations across groups but not necessarily independence within groups), "robust standard errors" were used in the logistic regression. These have the ability to relax the assumption of independence of the observations, i.e. they can produce "correct" standard errors (in the measurement sense) even if the observations are correlated . We chosTo test the goodness of fit, the Hosmer Lemeshaw goodness of fit statistics was applied to logistic regression model .The spread of data by pubertal stage is shown in Table The associations between back pain anywhere and the covariates expressed as odds ratios were: 2.9  for smoking and 0.7  for overweight.To our knowledge, this is the first time that the presence of back pain is studied in relation to pubertal stage. The results indicate that back problems and back pain might be related to puberty stage.th grade Finnish children was not found to be associated with self-reported low back pain [Two possibilities related to puberty spring to mind. First, the growth spurt initiated during puberty could be the initiating factor of back pain and back problems, as has been suggested by others ,12. For ack pain . ObviousWe found one study, in which low back- and neck/shoulder pain was studied in relation to whether the menarche started early, average or late in 14, 16 and 18 year old Finnish adolescents [Second, hormone-induced changes at puberty might have an effect on the attitudes to or perception of pain. We could find no information as to any evidence for or against such suppositions. Nonetheless, the perception of experimentally induced pain was noted to fluctuate across the menstrual cycle in females  and painWe decided not to include physical activity, age and height in the analysis although we have access to that information in our dataset. The reasons were that we have shown earlier that in this cohort objectively measured physical activity was not related to back pain , and preThe results should and have been interpreted with caution for two reasons. First, it is a cross sectional study and a secondary analysis. Our title: "Back pain reporting in young girls appears to be puberty-related", illustrates that the results derive from a hypothesis generating analysis rather than confirmatory analysis. To be able to perform a confirmatory analysis, a prospective study, including both genders from age 8 to 18 with frequent follow-ups, would be required. Second, when stratifying on the different spinal regions and puberty the number of subjects in the calculations gets small, and the odds ratios could therefore be inflated. But as the trend in the results is the same, in the analyses of back pain anywhere alone and of low back pain across the puberty stages, we believe that the association is true.Ideally, we should have made the same analyses on the boys, as data on these were collected. However, we found a systematic error, as the distribution of boys having started puberty was illogical, 85% being classified as having reached end of puberty, in comparison with only 43% of the girls. As girls generally start and end puberty before boys and the end of puberty in boys usually is around age 18, the data on puberty in boys could not be included in the study. It therefore now remains to find out if back pain reporting in boys is also linked with puberty and whether back pain is simply an inevitable aspect of growing up.8–10-yr old children and 14–16-yr old adolescents, attending state schools in Odense, Denmark, and their parents were invited to participate in a cardiovascular study.rd and 9th grades. Children were randomly selected using random number tables.Schools were stratified according to type , location , and socio-economic uptake of area. Proportional, two-stage cluster sample of children from each stratum. Primary units (clusters): schools; secondary units: children in 3Agreed to participate: 25/28 schools (89%) and 1020/1356 child-parent pairs (75%). Of these, 806 were included in the present study.The distribution of parental income and educational levels were representative of the national distribution .Questionnaire data were collected from parents, interviews conducted with children, and physical measurements made of children in a cross-sectional survey. The study was approved by the local ethics committee.Questionnaires were handled through teachers and by mail for non-responders. They contained: information on parental health, cardiovascular disease, cardiovascular risk factors, and socioeconomic status, plus information on children's birth weight and chronic diseases.Back pain interviews were conducted by NW and tested for face validity. Children were asked if they had any back or neck pain within the preceding month and whether the back pain had any consequences.Height, weight, body fatness and stage of puberty were measured but not all used in the present report.None of the variables was validated against a golden measure.Data entry was manual, checked by a second person. There was an additional verification and, if necessary, correction of outliers but none was removed from the data set.The author(s) declare that they have no competing interests.NW and CLY developed the aim and analytical approach for this paper. NW, LBA and KF were responsible for elements of the study design specific to this analysis. KF, NW and LBA obtained funding, co-ordinated and performed data collection. NW undertook the statistical analyses and NW and CLY wrote the first draft of the paper. All authors contributed to the final version.NW and CLY act as guarantors.The pre-publication history for this paper can be accessed here:


Incorporating evidence based medicine (EBM) into clinical practice requires clinicians to learn to efficiently gain access to clinical evidence and effectively appraise its validity. Even using current electronic systems, selecting literature-based data to solve a single patient-related problem can require more time than practicing physicians or residents can spare. Clinical librarians, as informationists, are uniquely suited to assist physicians in this endeavor.To improve support for evidence-based practice, we have developed a web-based EBM library consult service application (LCS). Librarians use the LCS system to provide full text evidence-based literature with critical appraisal in response to a clinical question asked by a remote physician. LCS uses an entirely Free/Open Source Software platform and will be released under a Free Software license. In the first year of the LCS project, the software was successfully developed and a reference implementation put into active use. Two years of evaluation of the clinical, educational, and attitudinal impact on physician-users and librarian staff are underway, and expected to lead to refinement and wide dissemination of the system.A web-based EBM library consult model may provide a useful way for informationists to assist clinicians, and is feasible to implement. Despite the growing availability of evidence-based medicine (EBM) training programs for practicing physicians and increasing emphasis on EBM in medical student and resident education, research indicates that even those clinicians who are most enthusiastic about EBM generally rely more on traditional information sources, such as consultation with respected colleagues, than on EBM-related sources . In reviEBM requires that clinicians learn new skills, including how to formulate questions about their patients that can be answered in the medical literature, how to search the clinical research literature for potentially relevant research reports, how to critically appraise the research design and analysis methods in order to determine the validity of reported results and their applicability to their patients, and how to use valid results appropriately in making clinical decisions. Such skills, the foundations of which lie in biostatistics, epidemiology, and library and information science, typically are not part of the armamentarium of medical school faculty and hence, are not easily promoted in medical students and residents. Even for those physicians who have acquired some skills and are committed to using them, access to the clinical evidence is far from easy, with searches often yielding either irrelevant citations or none at all . While uSince the creation, some 30 years ago, of the clinical medical librarianship program (CML) by Gertrude Lamb, several studies have demonstrated the effectiveness of an active partnership between clinicians and clinical librarians ,10. ClinIncreasingly, library authorities emphasize the importance of rethinking medical librarians' roles in the providing of medical information. Klein and Ross  call forIn an effort to actualize these recommendations, improve support for evidence-based practice, and increase physicians' use of EBM, a team of physicians, medical educators, programmers and librarians developed a web-based EBM library consult service application (LCS). The LCS system is designed to provide full text evidence-based literature with critical appraisal in response to a clinical question asked by a physician who may be at a remote or rural site. The first year of the LCS project focused on software development and reference implementation. During the upcoming two years, the service will be provided to two clinical departments at the University of Illinois at Chicago (UIC), and evaluated by its users using multiple methods. In addition, a second, separate LCS system will be implemented and evaluated at the University of Illinois at Chicago Peoria campus targeting rural community physicians.The idea of providing clinicians with a consult service focused on addressing clinical decisions with evidence has been practiced in other settings. Two particularly notable examples include the Clinical Decision Consultation Service at the New England Medical Center, which provides decision analysis consultations by physician-analysts to clinicians with a turnaround as fast as 24 hours for urgent cases , and theThe reference implementation of the LCS is at the Library of Health Sciences (LHS) at the UIC College of Medicine. The UIC College of Medicine is the largest medical school in the United States, and has a faculty of four thousand  at four locations across the state: Chicago, Peoria, Rockford, and Urbana-Champaign. Nearly 1300 medical students are educated each year.The primary clinical site for UIC is the University of Illinois Medical Center at Chicago, a large urban medical center that serves a socioeconomically diverse population. Approximately 300,000 outpatient visits are made each year. Patients are predominantly African-American (51%), Hispanic (24%), and White (21%), and represent a fairly uniform distribution of ages from newborn to over-65. Most patients are enrolled in HMOs or Medicaid.The University of Illinois Medical Center is connected to a high-speed university FDDI network with OC3 Internet connectivity. The primary information resources for clinicians at UIC are provided by LHS. LHS supports the College of Medicine's mission of teaching, research and service at its main campus in Chicago as well as at the three sites of the medical school located in Peoria, Rockford, and Urbana where site libraries are also staffed by health sciences librarians. The University Library currently provides access to over 20,000 electronic journals, 16,000 current serial titles and 1.9 million volumes. LHS serves as the Regional Medical Library for the ten-state Greater Midwest Region under a contract awarded by the U.S. National Library of Medicine.The reference implementation of LCS serves the residents and faculty of the Departments of Pediatrics and Family Medicine at the UIC College of Medicine. There are approximately 78 residents and 64 full-time physician faculty in these departments. The reference implementation is staffed by six clinical librarians and library residents at UIC.The basic design of the LCS is intended to mirror and enhance the way that clinicians and clinical librarians naturally interact. The clinician submits a query to the LCS using a dynamic web form that prompts for the elements of an answerable clinical question  Figures, 2, 3, 4The notified librarians are responsible for assigning the question to a librarian. Because any number of librarians can be notified when questions are received, LCS supports several different models of library staffing. For example, a single librarian can be designated to be notified of new questions, and given the responsibility of assigning each question to a suitable librarian, or all librarians can be notified of new questions, and any librarian can choose to answer. Once a librarian is assigned to a question, the librarian  is notified by email.The librarian may need clarification of the question by the clinician, which can take place either through LCS or without the mediation of LCS. The assigned librarian may submit a request for clarification through LCS, which is stored with the question and emailed to the clinician, or may contact the clinician directly by phone.The assigned librarian then answers the question by performing a search using library resources , and fills out a response form that mimics the "Critically Appraised Topic" (CAT) format recommended by Sauve, et al.  Figure LCS emails the completed response to the clinician, who can also retrieve it by logging into LCS. Clinicians are reminded (in the email and on login) to provide feedback and rate the quality of the answers they receive; once responses are rated, the interaction  is considered complete. LCS system procedures are illustrated in Figure For both practical and philosophical reasons, the system was designed to be licensed as free/open source software (FOSS) ,26 and tLinux was chosen because it runs on a variety of hardware platforms, providing a consistent environment. The reference hardware platforms include a Sun Ultra 5 workstation running Debian GNU/Linux 3.1 for SPARC and a Dell workstation running Debian GNU/Linux 3.1 for Intel x86 hardware .The Apache web server similarly is available for a wide range of operating systems. Although the LCS implementation uses Apache 1.3.33 and the MySQL 4.1 was chosRuby is a comThe LCS database uses a relatively simple relational structure. The primary database entities include users, questions, and responses Figure ; seconda® [The key interface design features for LCS include a familiar visual design that emphasizes usability, personalization of web pages, and online context-sensitive help. The primary interface consists of a navigation menu on the left side of the screen and one or more "blocks" of content on the right side of the screen that change as users select options from the navigation menu, a design similar to that employed by PUBMED®  By defauPersonalization is a critical element of the design, and serves to make the information load manageable for system users by clearly distinguishing between information targeted to the user and other information . On login, the service provides each user (librarian or physician) with a personal block, customized based on past interactions with the system. For example, a librarian sees a queue of queries assigned and unassigned. Similarly, physician users of the system receive a personalized list of their own pending queries. Users can also access responses to queries of others, with the questioner's identity masked.Online context-sensitive help is built into the system, and can be extended by the system administrator without knowledge of Ruby. Each page presented to users may have its own help, which is implemented using a Wiki-like collection of keywords associated with HTML pages.As discussed above, LCS is programmed primarily in the Ruby programming language, with database queries issued using Structured Query Language (SQL). Some web pages also use Javascript for browser detection or dynamic form generation. The Ruby source code is organized around several key objects that encapsulate the functionality of the database  and the interface , and associated methods that implement application logic . The Ruby PageTemplate library is employed throughout to separate the page display from the application logic and make it simpler to customize or modify the visual design; each web page, block, or email message generated by the system is represented by a template file that can be edited without knowledge of Ruby. This is particularly important in the early stages of system implementation, as system modification is expected in response to feedback from librarian and clinician users. It also makes it simpler for future installations of the system by others to customize the look of the system to fit local needs.Releases of the LCS software are available from the project's SourceForge project page .During the next two years of the project, the reference implementation of the LCS will be evaluated for feasibility, usefulness, and educational impact. In addition, a second LCS will be implemented and evaluated at the UIC Library of the Health Sciences – Peoria with a cohort of twenty rural community physicians.Feasibility of the service will be evaluated by documenting the processes of development, training, implementation, and evaluation. The documentation will consider each project phase individually and include a detailed log of the activities required during the phase and the resources required . This evaluation will be useful for establishing the replicability of the service at other sites and for estimating how the service might scale to support larger numbers of users.Usefulness measures include frequency and type of use, attitudes toward the service, evaluation of individual responses to questions, and a comparison of the quality of responses produced by the service to those produced by the physician-users themselves. The frequency of use of the service is measured and subtotaled on a monthly basis. Several relevant metrics are computed on a quarterly basis, such as the average daily question load per librarian, average time from question to response, and distribution of question types and sources of evidence returned.User and staff attitudes toward the consult service are assessed every six months using a locally-developed assessment instrument. The primary approach in this evaluation is within-subject, as we anticipate both substantial individual differences and, in the case of staff, small sample sizes. Changes in the attitude toward the consult service subscale will be modeled using hierarchical linear modeling of individual change parameters. This apThe value of the specific information provided by the service is rated by users at the time that they receive the information. On the web page that presents the consult results to physicians, they are asked to rate the relevance of the evidence received, the quality of the interpretation, the likelihood that the evidence will have an impact on the patient's care, and the likelihood that the evidence will affect how they treat patients in the future, using 7-point category rating scales. They are also offered the opportunity to provide open-ended feedback about the consult. These data will be examined every six months.Finally, an evaluation study is planned to provide convergent evidence about the value of the consult service in locating information. In this study, a representative subsample of the physician users will be asked to perform their own searches in response to answerable clinical questions (submitted by other users to the consult service within the last six months) and select the article(s) they would read to answer the question. Searching time required will be recorded, and the citations returned will be compared to those returned earlier by the consult service. Differences will be characterized qualitatively by the investigators. In addition, a subsample of responses by librarians will be compared to critical appraisals of the same articles by the investigators to evaluate and assure the quality of the librarian appraisals.Educational impact of the LCS on EBM skills and attitudes of both physicians and librarians will be evaluated regularly during the upcoming years of the project. Assessment tools have been developed to measure critical appraisal skills , as wellThe web-based Library Consult Service represents a natural evolution of the processes of evidence-based practice. Its release as, and reliance on, Free/Open Source Software offers administrators the freedom to modify the system to suit their needs and is likely to be cost-effective for experimentation by libraries. As an information system, it connects clinicians who have patient-oriented information needs with clinical librarians who have expertise in search and appraisal of the medical literature. It may also serve as an effective platform for EBM education and research on evidence-based clinical practice.Project name: EBM Library Consult ServiceProject home page: Operating system(s): LinuxProgramming language: RubyOther requirements: Ruby 1.8 with PageTemplate 2.1.6, MySQL 4.1 or higherLicense: GNU GPL version 2 or laterThe author(s) declare that they have no competing interests.AS conceived of the project, participated in its design and coordination, and drafted the manuscript. GM participated in the design of the system, performed system programming, and helped to draft the manuscript. All authors read and approved the final manuscript.The UIC LCS Investigators consist of Alan Schwartz, Ph.D. , Jordan Hupert, M.D. (Department of Pediatrics), Carol Scherrer, M.A.L.S. , Karen Connell, M.S. (Department of Family Medicine), Jerry Niederman, M.D., M.P.H. (Department of Pediatrics), and Josephine Dorsch, M.A.L.S. , all of whom meet the Vancouver criteria for authorship of this paper. Gregory Millam was responsible for the majority of the programming of the LCS itself and also co-authored this paper.The pre-publication history for this paper can be accessed here: