Diagnosed prevalence of Ehlers-Danlos syndrome and hypermobility spectrum disorder in Wales, UK: a national electronic cohort study and case–control comparison

Objectives To describe the epidemiology of diagnosed hypermobility spectrum disorder (HSD) and Ehlers-Danlos syndromes (EDS) using linked electronic medical records. To examine whether these conditions remain rare and primarily affect the musculoskeletal system. Design Nationwide linked electronic cohort and nested case–control study. Setting Routinely collected data from primary care and hospital admissions in Wales, UK. Participants People within the primary care or hospital data systems with a coded diagnosis of EDS or joint hypermobility syndrome (JHS) between 1 July 1990 and 30 June 2017. Main outcome measures Combined prevalence of JHS and EDS in Wales. Additional diagnosis and prescription data in those diagnosed with EDS or JHS compared with matched controls. Results We found 6021 individuals (men: 30%, women: 70%) with a diagnostic code of either EDS or JHS. This gives a diagnosed point prevalence of 194.2 per 100 000 in 2016/2017 or roughly 10 cases in a practice of 5000 patients. There was a pronounced gender difference of 8.5 years (95% CI: 7.70 to 9.22) in the mean age at diagnosis. EDS or JHS was not only associated with high odds for other musculoskeletal diagnoses and drug prescriptions but also with significantly higher odds of a diagnosis in other disease categories (eg, mental health, nervous and digestive systems) and higher odds of a prescription in most disease categories (eg, gastrointestinal and cardiovascular drugs) within the 12 months before and after the first recorded diagnosis. Conclusions EDS and JHS (since March 2017 classified as EDS or HSD) have historically been considered rare diseases only affecting the musculoskeletal system and soft tissues. These data demonstrate that both these assertions should be reconsidered

Active Children Through Individual Vouchers Evaluation: A Mixed-Method RCT

Introduction Physical activity declines in adolescence, especially among those in deprived areas. Research suggests this may result from accessibility barriers (e.g., cost and locality). The Active Children Through Individual Vouchers Evaluation RCT aimed to improve the fitness and heart health of teenagers in Wales with the help of teenagers who co-produced the study. Study design This study was a mixed-method RCT. Setting/participants Before data collection, which took place at baseline, 6 months, and 12 months for both arms, 7 schools were randomized by an external statistician (4 intervention schools, n=524; 3 control schools, n=385). Intervention The Active Children Through Individual Vouchers Evaluation intervention included provision of activity vouchers (£20 per month), a peer mentoring scheme, and support worker engagement for 12 months between January and December 2017. Data analysis occurred February–April 2018. Main outcome measures Data included measures of cardiovascular fitness, cardiovascular health (blood pressure and pulse wave analysis), motivation, and focus groups. Results The intervention showed a trend to improve the distance ran (primary outcome) and was significant in improving the likelihood of intervention teenagers being fit (OR=1.21, 95% CI=1.07, 1.38, p=0.002). There was a reduction in teenagers classified as having high blood pressure (secondary outcome) in the intervention group (baseline, 5.3% [28/524]; 12 months, 2.7% [14/524]). Data on where teenagers used vouchers and evidence from focus groups showed that teenagers wanted to access more unstructured, informal, and social activities in their local areas. Conclusions Active Children Through Individual Vouchers Evaluation identified methods that may have a positive impact on cardiovascular fitness, cardiovascular health, and perspectives of activity. Consulting with teenagers, empowering them, and providing more local opportunities for them to take part in activities that are fun, unstructured, and social could positively impact teenage physical activity

Internet-based randomised controlled trials for the evaluation of complementary and alternative medicines: probiotics in spondyloarthropathy

<b>Background</b> The clinical effectiveness of complementary and alternative medicines (CAMs) is widely debated because of a lack of clinical trials. The internet may provide an effective and economical approach for undertaking randomised controlled trials (RCTs) of low-risk interventions. We investigated whether the internet could be used to perform an internet-based RCT of a CAM fulfilling the revised CONSORT (Consolidated Standards of Reporting Trials) statement quality checklist for reporting of RCTs. A secondary aim was to examine the effect of probiotics compared to placebo in terms of well-being over 12 weeks.<p></p> <b>Methods</b> People aged ≥18 years with confirmed spondyloarthropathy living in the United Kingdom with internet access were invited to participate in an internet-based RCT of probiotic compared to placebo for improving well-being and bowel symptoms. The intervention was a probiotic containing 4 strains of live bacteria or identical placebo taken by mouth daily for 3 months. The primary outcome measure was the performance of the trial according to the revised CONSORT statement.<p></p> <b>Results</b> 147 people were randomised into the trial. The internet-based trial of the CAM fulfilled the revised CONSORT statement such as efficient blinding, allocation concealment, intention to treat analysis and flow of participants through the trial. Recruitment of the required number of participants was completed in 19 months. Sixty-five percent (96/147) completed the entire 3 months of the trial. The trial was low cost and demonstrated that in an intention to treat analysis, probiotics did not improve well-being or bowel symptoms.<p></p> <b>Conclusion</b> The internet-based RCT proved to be a successful and economical method for examining this CAM intervention. Recruitment, adherence and completion rate were all similar to those reported with conventional RCTs but at a fraction of the cost. Internet-based RCTs can fulfil all the criteria of the revised CONSORT statement and are an appropriate method for studying low-risk interventions

Protocol for a population-based Ankylosing Spondylitis (PAS) cohort in Wales

<p>Abstract</p> <p>Background</p> <p>To develop a population-based cohort of people with ankylosing spondylitis (AS) in Wales using (1) secondary care clinical datasets, (2) patient-derived questionnaire data and (3) routinely-collected information in order to examine disease history and the health economic cost of AS.</p> <p>Methods</p> <p>This data model will include and link (1) secondary care clinician datasets (i.e. electronic patient notes from the rheumatologist) (2) patient completed questionnaires (giving information on disease activity, medication, function, quality of life, work limitations and health service utilisation) and (3) a broad range of routinely collected data (including; GP records, in-patient hospital admission data, emergency department data, laboratory/pathology data and social services databases). The protocol involves the use of a unique and powerful data linkage system which allows datasets to be interlinked and to complement each other.</p> <p>Discussion</p> <p>This cohort can integrate patient supplied, primary and secondary care data into a unified data model. This can be used to study a range of issues such as; the true economic costs to the health care system and the patient, factors associated with the development of severe disease, long term adverse events of new and existing medication and to understand the disease history of this condition. It will benefit patients, clinicians and health care managers. This study forms a pilot project for the use of routine data/patient data linked cohorts for other chronic conditions.</p

HERALD (Health Economics using Routine Anonymised Linked Data)

&lt;b&gt;Background&lt;/b&gt; Health economic analysis traditionally relies on patient derived questionnaire data, routine datasets, and outcomes data from experimental randomised control trials and other clinical studies, which are generally used as stand-alone datasets. Herein, we outline the potential implications of linking these datasets to give one single joined up data-resource for health economic analysis.&lt;p&gt;&lt;/p&gt; &lt;b&gt;Method&lt;/b&gt; The linkage of individual level data from questionnaires with routinely-captured health care data allows the entire patient journey to be mapped both retrospectively and prospectively. We illustrate this with examples from an Ankylosing Spondylitis (AS) cohort by linking patient reported study dataset with the routinely collected general practitioner (GP) data, inpatient (IP) and outpatient (OP) datasets, and Accident and Emergency department data in Wales. The linked data system allows: (1) retrospective and prospective tracking of patient pathways through multiple healthcare facilities; (2) validation and clarification of patient-reported recall data, complementing the questionnaire/routine data information; (3) obtaining objective measure of the costs of chronic conditions for a longer time horizon, and during the pre-diagnosis period; (4) assessment of health service usage, referral histories, prescribed drugs and co-morbidities; and (5) profiling and stratification of patients relating to disease manifestation, lifestyles, co-morbidities, and associated costs.&lt;p&gt;&lt;/p&gt; &lt;b&gt;Results&lt;/b&gt; Using the GP data system we tracked about 183 AS patients retrospectively and prospectively from the date of questionnaire completion to gather the following information: (a) number of GP events; (b) presence of a GP 'drug' read codes; and (c) the presence of a GP 'diagnostic' read codes. We tracked 236 and 296 AS patients through the OP and IP data systems respectively to count the number of OP visits; and IP admissions and duration. The results are presented under several patient stratification schemes based on disease severity, functions, age, sex, and the onset of disease symptoms.&lt;p&gt;&lt;/p&gt; &lt;b&gt;Conclusion&lt;/b&gt; The linked data system offers unique opportunities for enhanced longitudinal health economic analysis not possible through the use of traditional isolated datasets. Additionally, this data linkage provides important information to improve diagnostic and referral pathways, and thus helps maximise clinical efficiency and efficiency in the use of resources

Educational Attainment at Age 10–11 Years Predicts Health Risk Behaviors and Injury Risk During Adolescence

Purpose: To examine the effect of educational attainment in primary school on later adolescent health. Methods: Education data attainments at age 7 and 11 were linked with (1) primary and secondary care injury consultation/admissions and (2) the Health Behaviour in School-aged Children survey. Cox regression was carried out to examine if attainment in primary school predicts time to injury in adolescence. Results: Pupils that achieve attainment at age 7 but not at age 11 (i.e., declining attainment over time in primary school) are more likely to have an injury during adolescence. These children are also more likely to self-report drinking in adolescence. Conclusions: Interventions aimed at children with declining attainment in primary school could help to improve adolescent health

Community led active schools programme (CLASP) exploring the implementation of health interventions in primary schools: headteachers’ perspectives

Background: Schools are repeatedly utilised as a key setting for health interventions. However, the translation of effective research findings to the school setting can be problematic. In order to improve effective translation of future interventions, it is imperative key challenges and facilitators of implementing health interventions be understood from a school's perspective. Methods: Nineteen semi-structured interviews were conducted in primary schools (headteachers n = 16, deputy headteacher n = 1, healthy school co-ordinator n = 2). Interviews were transcribed verbatim and analysed using thematic analysis. Results: The main challenges for schools in implementing health interventions were; government-led academic priorities, initiative overload, low autonomy for schools, lack of staff support, lack of facilities and resources, litigation risk and parental engagement. Recommendations to increase the application of interventions into the school setting included; better planning and organisation, greater collaboration with schools and external partners and elements addressing sustainability. Child-centred and cross-curricular approaches, inclusive whole school approaches and assurances to be supportive of the school ethos were also favoured for consideration. Conclusions: This work explores schools' perspectives regarding the implementation of health interventions and utilises these thoughts to create guidelines for developing future school-based interventions. Recommendations include the need to account for variability between school environments, staff and pupils. Interventions with an element of adaptability were preferred over the delivery of blanket fixed interventions. Involving schools in the developmental stage would add useful insights to ensure the interventions can be tailored to best suit each individual schools' needs and improve implementation.11 page(s