An evidence gap map of interventions for noncommunicable diseases and risk factors among children and adolescents.

Substance misuse, obesity, mental health conditions, type 1 diabetes, cancers, and cardiovascular and chronic respiratory diseases together account for 41% of disability-adjusted life years linked to noncommunicable diseases (NCDs) among children and adolescents worldwide. However, the evidence on risk factors and interventions for this age group is scarce. Here we searched four databases to generate an evidence gap map of existing interventions and research gaps for these risk factors and NCDs. We mapped 159 reviews with 2,611 primary studies; most (96.2%) were conducted in high-income countries, and only 100 studies (3.8%) were from low- and middle-income countries (LMICs). The efficacy of therapeutic interventions on biomarkers and adverse events for NCDs appears to be well evidenced. Interventions for mental health conditions appear to be moderately evidenced, while interventions for obesity and substance misuse appear to be moderate to very low evidenced. Priority areas for future research include evaluating digital health platforms to support primary NCD prevention and management, and evaluating the impact of policy changes on the prevalence of obesity and substance misuse. Our findings highlight the wide disparity of evidence between high-income countries and LMICs. There is an urgent need for increased, targeted financing to address the research gaps in LMICs.

Young Adults' Hopes Regarding Noncommunicable Disease (NCD) Prevention and Management After the SARS-CoV-2 Virus Pandemic: Reflections From the NCD Child #YouthCOVIDCHAT Campaign.

PURPOSE: The SARS-CoV-2 virus pandemic has left a massive global death toll in its wake. Associated restrictions, precautions and lockdowns have disrupted daily routines, which has been associated with social isolation and major health implications for the world's youth. This paper shares young adults' visions for life beyond the pandemic as it relates to the prevention and management of noncommunicable diseases (NCDs). METHODS: NCD Child hosted a global Twitter campaign for young adults, some of whom are living with NCDs, to express their reflections on life beyond the pandemic. Contributions were subjected to qualitative thematic analysis. RESULTS: 52 responses from the campaign described six main themes: Health system strengthening; Access to care; Issues of sustainability, including the environment and the economy; Human rights, equity, and social issues; Mental health, and NCD prevention. DISCUSSION: Young adults expressed optimism about postpandemic life and emphasized the importance of comprehensive intersectoral approaches to create resilient health systems.

A Model Predicting Healthcare Capacity Gaps For Alzheimer's Disease-Modifying Treatment in Canada.

BACKGROUND: Alzheimer's disease (AD) is experienced by > 600,000 Canadians. Disease-modifying therapies (DMTs) for earlier stages of disease are in development. Existing health system capacity constraints and the need for biomarker-driven diagnostics to confirm DMT eligibility are concerning. This study aimed to characterize the capacity gap related to early AD (eAD) treatment with DMTs in Canada. METHODS: A capacity model was developed to simulate the flow of a patient from screening to treatment for eAD to quantify the gap between available and required healthcare resources and qualify the bottlenecks restricting the patient journey at a provincial and national level. The model inputs (epidemiological, human resource, and clinical) were evidence-based, healthcare professional-, and patient advocate-informed. RESULTS: The model estimated that nationally < 2% of patients would have access to the required healthcare resources for treatment with a DMT. Eligibility assessment represented the step with the largest capacity gap across all provinces, with a wait list of about 382,000 Canadians one year following DMT introduction. The top three resource gaps included AD specialist time and positron emission tomography and magnetic resonance imaging exam slots. Sensitivity analysis showed that full reliance on cerebrospinal fluid for eligibility testing increased capacity for assessment by about 47,000 patients. CONCLUSION: This model highlights that the Canadian health system is critically under-resourced to diagnose, assess, and treat patients with eAD with DMT. It underscores an urgent need for national policy and provincial resource allocation to close the gap.

Why the trial researcher matters: Day-to-day work viewed through the lens of normalization process theory.

Researchers working in the field, the places where research-relevant activity happens, are essential to recruitment and data collection in randomised controlled trials (RCTs). This study aimed to understand the nature of this often invisible work. Data were generated through an RCT of a pharmacist-led medication management service for older people in care homes. The study was conducted over three years and employed seven Research Associates (RA) working in Scotland, Northern Ireland, and England. Weekly research team meetings and Programme Management Group meetings naturally generated 129 sets of minutes. This documentary data was supplemented with two end-of-study RA debriefing meetings. Data were coded to sort the work being done in the field, then deductively explored through the lens of Normalization Process Theory to enable a greater understanding of the depth, breadth and complexity of work carried out by these trial delivery RAs. Results indicate RAs helped stakeholders and participants make sense of the research, they built relationships with participants to support retention, operationalised complex data collection procedures and reflected on their own work contexts to reach agreement on changes to trial procedures. The debrief discussions enabled RAs to explore and reflect on experiences from the field which had affected their day-to-day work. The learning from the challenges faced in facilitating care home research may be useful to inform future research team preparation for complex interventions. Scrutinising these data sources through the lens of NPT enabled us to identify RAs as linchpins in the successful conduct of a complex RCT study.

Outcomes in Ankle Replacement Study (OARS).

Aims: The aim of this study was to capture 12-month outcomes from a representative multicentre cohort of patients undergoing total ankle arthroplasty (TAA), describe the pattern of patient-reported outcome measures (PROMs) at 12 months, and identify predictors of these outcome measures. Methods: Patients listed for a primary TAA at 19 NHS hospitals between February 2016 and October 2017 were eligible. PROMs data were collected preoperatively and at six and 12 months including: Manchester-Oxford Foot and Ankle Questionnaire (MOXFQ (foot and ankle)) and the EuroQol five-dimension five-level questionnaire (EQ-5D-5L). Radiological pre- and postoperative data included Kellgren-Lawrence score and implant position measurement. This was supplemented by data from the National Joint Registry through record linkage to determine: American Society of Anesthesiologists (ASA) grade at index procedure; indication for surgery, index ankle previous fracture; tibial hind foot alignment; additional surgery at the time of TAA; and implant type. Multivariate regression models assessed outcomes, and the relationship between MOXFQ and EQ-5D-5L outcomes, with patient characteristics. Results: Data from 238 patients were analyzed. There were significant improvements in MOXFQ and EQ-5D-5L among people who underwent TAA at six- and 12-month assessments compared with preoperative scores (p < 0.001). Most improvement occurred between preoperative and six months, with little further improvement at 12 months. A greater improvement in MOXFQ outcome postoperatively was associated with older age and more advanced radiological signs of ankle osteoarthritis at baseline. Conclusion: TAA significantly benefits patients with end-stage ankle disease. The lack of substantial further overall change between six and 12 months suggests that capturing PROMs at six months is sufficient to assess the success of the procedure. Older patients and those with advanced radiological disease had the greater gains. These outcome predictors can be used to counsel younger patients and those with earlier ankle disease on the expectations of TAA.

Availability and access to pediatric diabetes care: a global descriptive study.

A decade since the discovery of insulin, the increasing prevalence of type 1 diabetes mellitus (T1DM) has underscored the prevailing inequalities in the provision of essential care for T1DM worldwide. However, the details on the availability of insulin types and associated medical devices remain unclear. A cross-sectional electronic survey was distributed across a global network of pediatric societies under the umbrella of the International Pediatric Association (IPA). Access to and availability of pediatric diabetes care were investigated using standardized questions. Responses from 25 of 132 pediatric societies across six regions were included. Pediatric endocrinologists typically manage T1DM together with pediatricians or adult endocrinologists. Nonetheless, 24% of the respondents reported pediatricians to be the sole healthcare professionals. According to the respondents, the patients were either partially or completely responsible for payments of insulin (40%), A1C (24%), C-peptide (28%), and antibody testing for diagnosis (28%). Government support is generally available for insulin, but this was merely 20% for insulin pumps and 12% for continuous glucose monitors. There are considerable disparities in the access, availability, and affordability of diabetes testing, medications, and support between countries with significant out-of-pocket payments for care. Country- and region-specific improvements to national programs are necessary to achieve optimal pediatric diabetes care globally.

Evaluation of effectiveness and safety of pharmacist independent prescribers in care homes: cluster randomised controlled trial.

OBJECTIVE: To estimate the effectiveness, cost effectiveness (to be reported elsewhere), and safety of pharmacy independent prescribers in care homes. DESIGN: Cluster randomised controlled trial, with clusters based on triads of a pharmacist independent prescriber, a general practice, and one to three associated care homes. SETTING: Care homes across England, Scotland, and Northern Ireland, their associated general practices, and pharmacy independent prescribers, formed into triads. PARTICIPANTS: 49 triads and 882 residents were randomised. Participants were care home residents, aged ≥65 years, taking at least one prescribed drug, recruited to 20 residents/triad. INTERVENTION: Each pharmacy independent prescriber provided pharmaceutical care to approximately 20 residents across one to three care homes, with weekly visits over six months. Pharmacy independent prescribers developed a pharmaceutical care plan for each resident, did medicines reviews/reconciliation, trained staff, and supported with medicines related procedures, deprescribing, and authorisation of prescriptions. Participants in the control group received usual care. MAIN OUTCOMES MEASURES: The primary outcome was fall rate/person at six months analysed by intention to treat, adjusted for prognostic variables. Secondary outcomes included quality of life (EQ-5D by proxy), Barthel score, Drug Burden Index, hospital admissions, and mortality. Assuming a 21% reduction in falls, 880 residents were needed, allowing for 20% attrition. RESULTS: The average age of participants at study entry was 85 years; 70% were female. 697 falls (1.55 per resident) were recorded in the intervention group and 538 falls (1.26 per resident) in the control group at six months. The fall rate risk ratio for the intervention group compared with the control group was not significant (0.91, 95% confidence interval 0.66 to 1.26) after adjustment for all model covariates. Secondary outcomes were not significantly different between groups, with exception of the Drug Burden Index, which significantly favoured the intervention. A third (185/566; 32.7%) of pharmacy independent prescriber interventions involved medicines associated with falls. No adverse events or safety concerns were identified. CONCLUSIONS: Change in the primary outcome of falls was not significant. Limiting follow-up to six months combined with a small proportion of interventions predicted to affect falls may explain this. A significant reduction in the Drug Burden Index was realised and would be predicted to yield future clinical benefits for patients. This large trial of an intensive weekly pharmacist intervention with care home residents was also found to be safe and well received. TRIAL REGISTRATION: ISRCTN 17847169.

Effects of interactions between anthropogenic stressors and recurring perturbations on ecosystem resilience and collapse.

Insights into declines in ecosystem resilience and their causes and effects can inform preemptive action to avoid ecosystem collapse and loss of biodiversity, ecosystem services, and human well-being. Empirical studies of ecosystem collapse are rare and hampered by ecosystem complexity, nonlinear and lagged responses, and interactions across scales. We investigated how an anthropogenic stressor could diminish ecosystem resilience to a recurring perturbation by altering a critical ecosystem driver. We studied groundwater-dependent, peat-accumulating, fire-prone wetlands known as upland swamps in southeastern Australia. We hypothesized that underground mining (stressor) reduces resilience of these wetlands to landscape fires (perturbation) by diminishing groundwater, a key ecosystem driver. We monitored soil moisture as an indicator of ecosystem resilience during and after underground mining. After landscape fire, we compared responses of multiple state variables representing ecosystem structure, composition, and function in swamps within the mining footprint with unmined reference swamps. Soil moisture declined without recovery in swamps with mine subsidence (i.e., undermined), but was maintained in reference swamps over 8 years (effect size 1.8). Relative to burned reference swamps, burned undermined swamps showed greater loss of peat via substrate combustion; reduced cover, height, and biomass of regenerating vegetation; reduced postfire plant species richness and abundance; altered plant species composition; increased mortality rates of woody plants; reduced postfire seedling recruitment; and extirpation of a hydrophilic animal. Undermined swamps therefore showed strong symptoms of postfire ecosystem collapse, whereas reference swamps regenerated vigorously. We found that an anthropogenic stressor diminished the resilience of an ecosystem to recurring perturbations, predisposing it to collapse. Avoidance of ecosystem collapse hinges on early diagnosis of mechanisms and preventative risk reduction. It may be possible to delay or ameliorate symptoms of collapse or to restore resilience, but the latter appears unlikely in our study system due to fundamental alteration of a critical ecosystem driver. Efectos de las interacciones entre los estresantes antropogénicos y las perturbaciones recurrentes sobre la resiliencia y el colapso de los ecosistemas.

La comprensión de la declinación en la resiliencia de los ecosistemas y sus causas y efectos puede orientar las acciones preventivas para evitar el colapso ecosistémico y la pérdida de biodiversidad, servicios ambientales y bienestar humano. Los estudios empíricos del colapso ecosistémico son escasos y se enfrentan a obstáculos como la complejidad del ecosistema, respuestas rezagadas y no lineales e interacciones entre las escalas. Investigamos cómo un estresante antropogénico podría reducir la resiliencia del ecosistema a una perturbación recurrente mediante la alteración de un causante importante. Estudiamos los humedales dependientes de aguas subterráneas que acumulan turbas y son propicios a incendios conocidos como pantanos de tierras altas en el sureste de Australia. Nuestra hipótesis fue que la minería subterránea (estresante) reduce la resiliencia de estos humedales a incendios (perturbación) al disminuir el agua subterránea, un causante clave para el ecosistema. Monitoreamos la humedad del suelo como un indicador de la resiliencia del ecosistema durante y después de la minería subterránea. Después de los incendios, comparamos la respuesta de múltiples variables de estado que representaban la estructura, composición y función del ecosistema en los pantanos dentro de la huella minera con los pantanos referenciales sin minería. La humedad del suelo declinó sin recuperación en los pantanos con hundimientos mineros (es decir, socavones) pero se mantuvo en los pantanos referenciales durante ocho años (tamaño del efecto: 1.8). En relación a los pantanos referenciales incendiados, los pantanos con socavones e incendios mostraron una mayor pérdida de turba mediante la combustión del sustrato; reducción en la cobertura, altura y regeneración de biomasa de la vegetación; reducción en la riqueza y abundancia de especies vegetales post incendio; alteraciones en la composición de especies vegetales; incremento en la mortalidad de las plantas leñosas; reducción en el reclutamiento post incendio de plántulas; y la extirpación de un animal hidrofílico. Por lo tanto, los pantanos con socavones mostraron síntomas fuertes de un colapso ecosistémico post incendio, mientras que los pantanos referenciales se regeneraron vigorosamente. Descubrimos que los estresantes antropogénicos redujeron la resiliencia de un ecosistema a perturbaciones recurrentes, lo que lo predispone al colapso. La eliminación de este colapso depende de un diagnóstico temprano de mecanismos y reducción del riesgo preventivo. Puede ser posible retardar o mitigar los síntomas del colapso o restaurar la resiliencia, aunque lo último parece ser improbable en nuestro sistema de estudio debido a la alteración fundamental de un causante importante del ecosistema.

English care home staff morale and preparedness during the COVID pandemic: A longitudinal analysis.

BACKGROUND: Staff actions to prevent infection introduction and transmission in long-term care facilities (LTCFs) were key to reducing morbidity and mortality from COVID-19. Implementing infection control measures (ICMs) requires training, adherence and complex decision making while trying to deliver high quality care. We surveyed LTCF staff in England about their preparedness and morale at 3 timepoints during the COVID-19 epidemic. METHODS: Online structured survey targeted at LTCF workers (any role) administered at 3 timepoints (November 2020-January 2021; August-November 2021; March-May 2022). Narrative summary of answers, narrative and statistical summary (proportionality with Pearson's chi-square or Fisher's Exact Test) of possible differences in answers between waves. RESULTS: Across all 3 survey waves, 387 responses were received. Morale, attitudes towards working environment and perception about colleague collaboration were mostly positive at all survey points. Infection control training was perceived as adequate. Staff felt mostly positive emotions at work. The working environment remained challenging. Masks were the single form of PPE most consistently used; eye protection the least used. Mask-wearing was linked to poorer communication and resident discomfort as well as mild negative health impacts on many staff, such as dehydration and adverse skin reactions. Hand sanitizer caused skin irritation. CONCUSIONS: Staff morale and working practices were generally good even though the working environment provided many new challenges that did not exist pre-pandemic.

Neuropathic-like Pain in Fibrous Dysplasia/McCune-Albright Syndrome.

CONTEXT: Pain is a major symptom in adults with fibrous dysplasia/McCune-Albright syndrome (FD/MAS) and response to current treatments, including bisphosphonates and standard analgesics (nonsteroidal anti-inflammatory drugs and opiates) is unpredictable. No studies have explored whether the type of pain is variable in this patient group. OBJECTIVE: To determine the frequency of neuropathic-like pain in patients with FD/MAS. DESIGN: Retrospective, dual registry study. SETTING: Community. PATIENTS: FD/MAS online registries: the US-based Familial Dysautonomia Foundation (FDF) and the UK-based Rare and Undiagnosed Diseases (RUDY) study. INTERVENTION: Subjects completed questionnaires to evaluate the presence of features of neuropathic-like pain (painDETECT) and the impact on sleep quality (Pittsburgh Sleep Quality Index) and mental health (Hospital Anxiety and Depression Scale). Descriptive statistics were used to characterize the prevalence and associated burden of neuropathic-like pain. MAIN OUTCOME MEASURES: Incidence of neuropathic, nociceptive, and unclear pain. RESULTS: Of 249 participants, one third experienced neuropathic-like pain. This group had statistically significantly (P < 0.001) worse mental well-being and sleep in comparison to those with predominately nociceptive pain. CONCLUSIONS: Neuropathic-like pain is common in patients with FD/MAS and associated with worse quality of life. Evaluation of pain in patients with FD/MAS should include assessment of neuropathic-like pain to guide personalized approaches to treatment and inform future research.

Dementia and sexuality in long-term care: Incompatible bedfellows?

Despite the recognized benefits of sexual expression and its importance in the lives of people living with dementia, research demonstrates that there are multiple barriers to its positive expression (e.g., expression that is pleasurable and free of coercion, discrimination, and violence) in RLTC homes. These barriers constitute a form of discrimination based on age and ability, and violate the rights of persons living with dementia to dignity, autonomy, and participation in everyday life and society. Drawing on a human rights approach to dementia and sexual expression, we explored the experiences of diverse professionals, family members, and persons living with dementia with explicit attention to the ways in which macro-level dynamics are influencing the support, or lack thereof, for sexual expression at the micro level. Focus groups and in-depth interviews were conducted with 27 participants, and the collected data were analyzed thematically. While all participants acknowledged that intimacy and sexual expression of persons living with dementia should be supported, rarely is such expression supported in practice. Micro-level factors included negative attitudes of professionals toward sexual expression by persons living with dementia, their discomfort with facilitating intimacy and sexual expression in the context of their professional roles, their anxieties regarding potential negative reactions from family members, and concerns about sanctions for failing to prevent abuse. In our analysis, we importantly trace these micro-level factors to macro-level factors. The latter include the cultural stigma associated with dementia, ageism, ableism, and erotophobia, all of which are reproduced in, and reinforced by, professionals' education, as well as legal and professional standards that exclusively focus on managing and safeguarding residents from abuse. Our analysis demonstrates a complexity that has enormous potential to inform future research that is critically needed for the development of educational initiatives and to promote policy changes in this area.

Clinical and biomechanical factors associated with falls and rheumatoid arthritis: baseline cohort with longitudinal nested case-control study.

OBJECTIVE: To identify the clinical and biomechanical characteristics associated with falls in people with RA. METHODS: A total of 436 people ≥60 years of age with RA completed a 1 year prospective survey of falls in the UK. At baseline, questionnaires recorded data including personal and medical history, pain and fatigue scores, health-related quality of life (HRQoL), physical activity and medication history. The occurrence of falls wasmonitored prospectively over 12 months by monthly self-reporting. A nested sample of 30 fallers (defined as the report of one or more falls in 12 months) and 30 non-fallers was evaluated to assess joint range of motion (ROM), muscle strength and gait parameters. Multivariate regression analyses were undertaken to determine variables associated with falling. RESULTS: Compared with non-fallers (n = 236), fallers (n = 200) were older (P = 0.05), less likely to be married (P = 0.03), had higher pain scores (P < 0.01), experienced more frequent dizziness (P < 0.01), were frequently taking psychotropic medications (P = 0.02) and reported lower HRQoL (P = 0.02). Among those who underwent gait laboratory assessments, compared with non-fallers, fallers showed a greater anteroposterior (AP; P = 0.03) and medial-lateral (ML) sway range (P = 0.02) and reduced isokinetic peak torque and isometric strength at 60° knee flexion (P = 0.03). Fallers also showed shorter stride length (P = 0.04), shorter double support time (P = 0.04) and reduced percentage time in swing phase (P = 0.02) and in knee range of motion through the gait cycle (P < 0.01). CONCLUSION: People with RA have distinct clinical and biomechanical characteristics that place them at increased risk of falling. Assessment for these factors may be important to offer more targeted rehabilitation interventions.

A century past the discovery of insulin: global progress and challenges for type 1 diabetes among children and adolescents in low-income and middle-income countries.

Type 1 diabetes is on the rise globally; however, the burden of mortality remains disproportionate in low-income and middle-income countries (LMICs). As 2021 marks 100 years since the discovery of insulin, we revisit progress, global burden of type 1 diabetes trends, and understanding of the pathogenesis and management practices related to the disease. Despite much progress, inequities in access and availability of insulin formulations persist and are reflected in differences in survival and morbidity patterns related to the disease. Some of these inequities have also been exacerbated by health-system challenges during the COVID-19 pandemic. There is a clear opportunity to improve access to insulin and related essential technologies for improved management of type 1 diabetes in LMICs, especially as a part of universal health coverage. These improvements will require concerted action and investments in human resources, community engagement, and education for the timely diagnosis and management of type 1 diabetes, as well as adequate health-care financing. Further research in LMICs, especially those in Africa, is needed to improve our understanding of the burden, risk factors, and implementation strategies for managing type 1 diabetes.

Awakening Canadians to ageism: a study protocol.

BACKGROUND: Making fun of growing older is considered socially acceptable, yet ageist humour reinforces negative stereotypes that growing old is linked with physical and mental deterioration, dependence, and less social value. Such stereotypes and discrimination affect the wellbeing of older people, the largest demographic of Canadians. While ageism extends throughout professions and social institutions, we expect nurses-the largest and most trusted group of healthcare professionals-to provide non-ageist care to older people. Unfortunately, nurses working with older people often embrace ageist beliefs and nursing education programs do not address sufficient anti-ageism content despite gerontological nursing standards and competencies. METHODS: To raise awareness of ageism in Canada, this quasi-experimental study will be supported by partnerships between older Canadians, advocacy organizations, and academic gerontological experts which will serve as an advisory group. The study, guided by social learning theory, will unfold in two parts. In Phase 1, we will use student nurses as a test case to determine if negative stereotypes and ageist perceptions can be addressed through three innovative e-learning activities. The activities employ gamification, videos, and simulations to: (1) provide accurate general information about older people, (2) model management of responsive behaviours in older people with cognitive impairment, and (3) dispel negative stereotypes about older people as dependent and incontinent. In Phase 2, the test case findings will be shared with the advisory group to develop a range of knowledge mobilization strategies to dispel ageism among healthcare professionals and the public. We will implement key short term strategies. DISCUSSION: Findings will generate knowledge on the effectiveness of the e-learning activities in improving student nurses' perceptions about older people. The e-learning learning activities will help student nurses acquire much-needed gerontological knowledge and skills. The strength of this project is in its plan to engage a wide array of stakeholders who will mobilize the phase I findings and advocate for positive perspectives and accurate knowledge about aging-older Canadians, partner organizations (Canadian Gerontological Nurses Association, CanAge, AgeWell), and gerontological experts.

Rare musculoskeletal diseases in adults: a research priority setting partnership with the James Lind Alliance.

BACKGROUND: Osteogenesis imperfecta, fibrous dysplasia/McCune-Albright syndrome and X-linked hypophosphatemia are three rare musculoskeletal diseases characterised by bone deformities, frequent fractures and pain. Little high-quality research exists on appropriate treatment and long-term management of these conditions in adults. This is further worsened by limited research funding in rare diseases and a general mismatch between the existing research priorities and those of the patients. This partnership adopted the James Lind Alliance approach to identify the top 10 research priorities for rare musculoskeletal diseases in adults through joint patient, carer and healthcare professional collaboration. RESULTS: The initial survey for question collection recruited 198 respondents, submitting a total of 988 questions. 77% of the respondents were patients with a rare musculoskeletal disease. Following out-of-scope question exclusion, repeating query grouping and scientific literature check for answers, 39 questions on treatment and long-term management remained. In the second public survey, 220 respondents, of whom 85% were patients with a rare musculoskeletal disease, their carers, relatives or friends, prioritised these uncertainties, which allowed selection of the top 25. In the last stage, patients, carers and healthcare professionals gathered for a priority setting workshop to reach a consensus on the final top 10 research priorities. These focus on the uncertainties surrounding appropriate treatment and holistic long-term disease management, highlighting several aspects indirect to abnormal bone metabolism, such as extra-skeletal symptoms, psychological care of both patients and their families and disease course through ageing. CONCLUSIONS: This James Lind Alliance priority setting partnership is the first to investigate rare bone diseases. The priorities identified here were developed jointly by patients, carers and healthcare professionals. We encourage researchers, funding bodies and other stakeholders to use these priorities in guiding future research for those affected by rare musculoskeletal disorders.

Understanding transitional care programmes for older adults who experience delayed discharge: a scoping review protocol.

INTRODUCTION: Many hospitalised older adults experience delayed discharges due to increased postacute health and social support needs. Transitional care programmes (TCPs) provide short-term care to these patients to prepare them for transfer to nursing homes or back to the community with supports. There are knowledge gaps related to the development, implementation and evaluation of TCPs. The aims of this scoping review (ScR) are to identify the characteristics of older patients served by TCPs; criteria for transfer, components and services provided by TCPs; and outcomes used to evaluate TCPs. METHODS AND ANALYSIS: The study involves six-step ScR and is informed by a collaborative/participatory approach whereby stakeholders engage in the development of the research questions, identification of literature, data abstraction and synthesis; and participation in consultation workshop. The search for scientific literature will be done in the Medline, PsychINFO, Emcare and CINAHL databases; as well, policies and reports that examined models of transitional care and the outcomes used to evaluate them will be reviewed. Records will be selected if they involve community dwelling older adults aged 65 years or older, or indigenous persons 45 years or older; and presented in English, French, Dutch and German languages. Records will be screened, reviewed and abstracted by two independent reviewers. Extracted data will be analysed using descriptive statistics and a narrative analysis, and organised according to Donabedian's model of structure (characteristics of older adults experiencing delayed discharge and served by TCPs), process (TCP components and services) and outcome. ETHICS AND DISSEMINATION: This ScR does not require ethics approval. Dissemination activities include integrated knowledge translation (KT) (consultation with stakeholders throughout the study) and end-of-grant KT strategies (presentations at national and international conferences; and publication in peer-reviewed interdisciplinary journal).

"It's my own fault": Accounts and consequences of falling when living with rheumatoid arthritis.

INTRODUCTION: Rheumatoid arthritis (RA) leads to biomechanical joint changes, which increases the risk of falling. The consequence of falling may be physical injury. However, the psychological consequences, including the fear of falling, can be equally important. METHODS: Participants were recruited from a larger prospective study which explored the incidence of falls in people with RA. Purposive sampling considered age, sex, time since diagnosis and fall history. The recruitment site was a regional hospital. Data were collected from semi-structured qualitative interviews and, after each fall, brief telephone interviews. Thematic analysis methods were used to investigate the psychological and social impact of falling in people with RA. RESULTS: Twelve participants were interviewed (aged 64-85, mean 74 years; six had fallen between one and 23 times, and six had no reported falls in last 12 months). Data were supplemented with telephone notes from 287 post-fall telephone calls. Three themes were developed: (i) the falls imaginary illustrates that the fear of falling is not dependent on experience; (ii) agentic risk management reports on the ways people self-manage and display resilience when at risk of falling; (iii) the absence of the health professional explores the ways in which people reported being unsupported by healthcare services. CONCLUSION: Fear of falling when living with RA is tangible in those who have and have not fallen. This fear may limit opportunities for full participation in life. However, some people display personal resourcefulness, continuing to live purposeful lives. Understanding personal responses to falling will support the development of community interventions specific to this high-risk group.

The effectiveness of a multidisciplinary intervention strategy for the treatment of symptomatic joint hypermobility in childhood: a randomised, single Centre parallel group trial (The Bendy Study).

INTRODUCTION: Joint hypermobility is common in childhood and can be associated with musculoskeletal pain and dysfunction. Current management is delivered by a multidisciplinary team, but evidence of effectiveness is limited. This clinical trial aimed to determine whether a structured multidisciplinary, multisite intervention resulted in improved clinical outcomes compared with standard care. METHOD: A prospective randomised, single centre parallel group trial comparing an 8-week individualised multidisciplinary intervention programme (bespoke physiotherapy and occupational therapy in the clinical, home and school environment) with current standard management (advice, information and therapy referral if deemed necessary). The primary endpoint of the study was between group difference in child reported pain from baseline to 12 months as assessed using the Wong Baker faces pain scale. Secondary endpoints were parent reported pain (100 mm visual analogue scale), parent reported function (child health assessment questionnaire), child reported quality of life (child health utility 9-dimensional assessment), coordination (movement assessment battery for children version 2) and grip strength (handheld dynamometer). RESULTS: 119 children aged 5 to 16 years, with symptomatic hypermobility were randomised to receive an individualised multidisciplinary intervention (I) (n = 59) or standard management (S) (n = 60). Of these, 105 completed follow up at 12 months. No additional significant benefit could be shown from the intervention compared to standard management. However, there was a statistically significant improvement in child and parent reported pain, coordination and grip strength in both groups. The response was independent of the degree of hypermobility. CONCLUSION: This is the first randomised controlled trial to compare a structured multidisciplinary, multisite intervention with standard care in symptomatic childhood hypermobility. For the majority, the provision of education and positive interventions aimed at promoting healthy exercise and self-management was associated with significant benefit without the need for more complex interventions. TRIAL REGISTRATION: The trial was registered prospectively with the national database at the Clinical Research Network (UKCRN Portfolio 9366). The trial was registered retrospectively with ISRCTN ( ISRCTN86573140 ).

The severity of ankylosing spondylitis and responses to anti-tumour necrosis factor biologics are not influenced by the tumour necrosis factor receptor polymorphism incriminated in multiple sclerosis.

Genetic polymorphism (rs1800693) of TNFRSF1A (type 1 tumour necrosis factor receptor) encodes a potentially anti-inflammatory soluble truncated form of the p55 receptor, which is associated with predisposition to multiple sclerosis but protection against ankylosing spondylitis (AS). We analysed 2917 UK Caucasian cases by linear and logistic regression for associations of rs1800693 with disease severity assessed by the Bath Ankylosing Spondylitis measures of disease activity and function (BASDAI, BAS-G and BASFI) and/or responses to anti-TNF therapy. In contrast to predictions, rs1800693 GG homozygotes actually had significantly worse BASDAI (mean 4.2, 95% CI: 4-4.5) than AA homozygotes (mean 3.8, 95% CI: 3.7-4) in both the unadjusted (difference = 0.4, p = 0.006) and adjusted analyses (difference = 0.2-0.5, p = 0.002-0.04 depending on the adjustment model). We found no evidence that rs1900693 predicted functional status (BASFI) or global disease scores (BAS-G), and it exerted no influence on either the intention to treat with or efficacy of anti-TNF treatment.