Correction: Currie et al. Reality Check 2: The Cost-Effectiveness of Policy Disallowing Body Checking in Non-Elite 13- to 14-Year-Old Ice Hockey Players. Int. J. Environ. Res. Public Health 2021, 18, 6322.

There was an error in our original publication [...].

Quantifying hospital-associated costs, and accompanying travel costs and productivity losses, before and after withdrawing tumour necrosis factor-alfa inhibitors in juvenile idiopathic arthritis.

OBJECTIVE: To quantify differences in hospital-associated costs, and accompanying travel costs and productivity losses, before and after withdrawing TNFi in JIA patients. METHODS: Retrospective analysis of prospectively collected data from electronic medical records of paediatric JIA patients treated with TNFi, which were either immediately discontinued, spaced (increased treatment interval) or tapered (reduced subsequent doses). Costs of hospital-associated resource use (consultations, medication, radiology procedures, laboratory testing, procedures under general anaesthesia, hospitalisation) and associated travel costs and productivity losses were quantified during clinically inactive disease until TNFi withdrawal (pre-withdrawal period) and compared with costs during the first and second year after withdrawal initiation (first and second year post-withdrawal). RESULTS: Fifty-six patients were included of whom 26 immediately discontinued TNFi, 30 spaced and zero tapered. Mean annual costs were €9,165/patient on active treatment (pre-withdrawal) and decreased significantly to €5,063/patient (-44.8%) and €6,569/patient (-28.3%) in the first and second year post-withdrawal, respectively (p< 0.05). Of these total annual costs, travel costs plus productivity losses were €834/patient, €1,180/patient, and €1,320/patient, in the three periods respectively. Medication comprised 80.7%, 61.5% and 72.4% of total annual costs in the pre-withdrawal, first, and second year post-withdrawal period, respectively. CONCLUSION: In the first two years after initiating withdrawal, the total annual costs are decreased compared with the pre-withdrawal period. However, cost reductions were lower in the second year compared with the first year post-withdrawal, primarily due to restarting or intensifying biologics. To support biologic withdraw decisions, future research should assess the full long-term societal cost impacts, and include all biologics.

Are We Capturing the Socioeconomic Burden of Rare Genetic Disease? A Scoping Review of Economic Evaluations and Cost-of-Illness Studies.

BACKGROUND AND OBJECTIVES: Rare diseases have a significant impact on patients, families, the health system, and society. Measuring the socioeconomic burden is crucial to valuing interventions for rare diseases. Healthcare system costs are significant, but so are costs to other government sectors, patients, families, and society. To understand the breadth of costs captured in rare disease studies, we examined the cost categories and elements of socioeconomic burden captured in published studies. METHODS: A scoping review was conducted using five electronic databases to identify English language economic evaluations and cost-of-illness studies of interventions for rare diseases (2011-21). We mapped costs using a previously developed evidence-informed framework of socioeconomic burden costs for rare disease. RESULTS: Of 4890 studies identified, 48 economic evaluations and 22 cost-of-illness studies were included. While 18/22 cost-of-illness studies utilized a societal perspective, only 7/48 economic evaluations incorporated societal costs. Most reported cost categories related to medical costs, with medication and hospitalizations being the most common elements for both study designs. Costs borne by patients, families, and society were reported less among economic evaluations than cost-of-illness studies. These included: productivity (10% vs 77%), travel/accommodation (6% vs 68%), government benefits (4% vs 18%), and family impacts (0% vs 50%). CONCLUSIONS: Contrary to cost-of-illness analyses, most of the included economic evaluations did not account for the hidden burden of rare diseases, that is, costs borne by patients, families, and societies. Including these types of costs in future studies would provide a more comprehensive picture of the burden of disease, providing empirical data to inform how we value and make decisions regarding rare disease interventions, health policy, and resource allocation.

Withdrawing biologics in non-systemic JIA: what matters to pediatric rheumatologists?

OBJECTIVE: Approximately one third of children with JIA receive biologic therapy, but evidence on biologic therapy withdrawal is lacking. This study aims to increase our understanding of whether and when pediatric rheumatologists postpone a decision to withdraw biologic therapy in children with clinically inactive non-systemic JIA. METHODS: A survey containing questions about background characteristics, treatment patterns, minimum treatment time with biologic therapy, and 16 different patient vignettes, was distributed among 83 pediatric rheumatologists in Canada and the Netherlands. For each vignette, respondents were asked whether they would withdraw biologic therapy at their minimum treatment time, and if not, how long they would continue biologic therapy. Statistical analysis included descriptive statistics, logistic and interval regression analysis. RESULTS: Thirty-three pediatric rheumatologists completed the survey (40% response rate). Pediatric rheumatologists are most likely to postpone the decision to withdraw biologic therapy when the child and/or parents express a preference for continuation (OR 6.3; p < 0.001), in case of a flare in the current treatment period (OR 3.9; p = 0.001), and in case of uveitis in the current treatment period (OR 3.9; p < 0.001). On average, biologic therapy withdrawal is initiated 6.7 months later when the child or parent prefer to continue treatment. CONCLUSION: Patient's and parents' preferences were the strongest driver of a decision to postpone biologic therapy withdrawal in children with clinically inactive non-systemic JIA and prolongs treatment duration. These findings highlight the potential benefit of a tool to support pediatric rheumatologists, patients and parents in decision making, and can help inform its design.

Barriers experienced by families new to Alberta, Canada when accessing routine-childhood vaccinations.

BACKGROUND: As Canada and other high-income countries continue to welcome newcomers, we aimed to 1) understand newcomer parents' attitudes towards routine-childhood vaccinations (RCVs), and 2) identify barriers newcomer parents face when accessing RCVs in Alberta, Canada. METHODS: Between July 6th-August 31st, 2022, we recruited participants from Alberta, Canada to participate in moderated focus group discussions. Inclusion criteria included parents who had lived in Canada for < 5 years with children < 18 years old. Focus groups were transcribed verbatim and analyzed using content and deductive thematic analysis. The capability opportunity motivation behaviour model was used as our conceptual framework. RESULTS: Four virtual and three in-person focus groups were conducted with 47 participants. Overall, parents were motivated and willing to vaccinate their children but experienced several barriers related to their capability and opportunity to access RCVs. Five main themes emerged: 1) lack of reputable information about RCVs, 2) language barriers when looking for information and asking questions about RCVs, 3) lack of access to a primary care provider (PCP), 4) lack of affordable and convenient transportation options, and 5) due to the COVID-19 pandemic, lack of available vaccine appointments. Several minor themes were also identified and included barriers such as lack of 1) childcare, vaccine record sharing, PCP follow-up. CONCLUSIONS: Our findings highlight that several barriers faced by newcomer families ultimately stem from issues related to accessing information about RCVs and the challenges families face once at vaccination clinics, highlighting opportunities for health systems to better support newcomers in accessing RCVs.

What matters most to pediatric rheumatologists in deciding whether to discontinue biologics in a child with juvenile idiopathic arthritis? A best-worst scaling survey.

OBJECTIVES: Care for JIA patients has been transformed in the biologics era; however, biologics carry important (although rare) risks and are costly. Flares after biological withdrawal are seen frequently, yet there is little clinical guidance to identify which patients in clinical remission can safely have their biologic discontinued (by stopping or tapering). We examined what characteristics of the child or their context are important to pediatric rheumatologists when making the decision to discuss withdrawal of biologics. METHODS: We conducted a survey including a best-worst scaling (BWS) exercise in pediatric rheumatologists who are part of the UCAN CAN-DU network to assess the relative importance of 14 previously identified characteristics. A balanced incomplete block design was used to generate choice tasks. Respondents evaluated 14 choice sets of 5 characteristics of a child with JIA and identified for each set which was the most and least important in the decision to offer withdrawal. Results were analyzed using conditional logit regression. RESULTS: Fifty-one (out of 79) pediatric rheumatologists participated (response rate 65%). The three most important characteristics were how challenging it was to achieve remission, history of established joint damage, and time spent in remission. The three least important characteristics were history of temporomandibular joint involvement, accessibility of biologics, and the patient's age. CONCLUSIONS: These findings give quantitative insight about factors important to pediatric rheumatologists' decision-making about biologic withdrawal. In addition to high quality clinical evidence, further research is needed to understand the perspective of patients and families to inform shared decision-making about biologic withdrawal for JIA patients with clinically inactive disease. Key Points ● What is already known on this topic-there is limited clinical guidance for pediatric rheumatologists in making decisions about biologic withdrawal for patients with juvenile idiopathic arthritis who are in clinical remission. ● What this study adds-this study quantitatively examined what characteristic of the child in clinical remission, or of their context, are most important to pediatric rheumatologists in deciding whether to offer withdrawal of biologics. ● How this study might affect research, practice or policy-understanding of these characteristics can provide useful information to other pediatric rheumatologists in making their decisions, and may guide areas to focus on for future research.

Developing a Framework of Cost Elements of Socioeconomic Burden of Rare Disease: A Scoping Review.

BACKGROUND AND OBJECTIVE: Rare diseases place a significant burden on patients, families, the healthcare system, and society. Evidence on the socioeconomic burden of rare disease is limited and mostly reflects diseases where treatments are available. We developed a framework encompassing recommended cost elements for studies of the socioeconomic burden of rare diseases. METHODS: A scoping review, conducted in five databases (Cochrane Library, EconLit, Embase, MEDLINE, and APA PsycINFO), identified English language publications from 2000 to 2021 presenting frameworks developed for determining, measuring or valuing costs for rare or chronic diseases. Cost elements were extracted and used to develop a literature-informed framework. Structured feedback was gathered from experts in rare diseases, health economics/health services, and policy research to revise the framework. RESULTS: Of 2990 records identified, eight papers were included and informed our preliminary framework; three focused on rare disease and five on chronic disease. Following expert input, we developed a framework consisting of nine cost categories (inpatient, outpatient, community, healthcare products/goods, productivity/education, travel/accommodation, government benefits, family impacts, and other), with several cost elements within each category. Our framework includes unique costs, added from the expert feedback, including genetic testing to inform treatment, use of private laboratories or out-of-country testing, family involvement in foundations and organizations, and advocacy costs for special access programs. CONCLUSIONS: Our work is the first to identify a comprehensive list of cost elements for rare disease for use by researchers and policy makers to fully capture socioeconomic burden. Use of the framework will increase the quality and comparability of future studies. Future work should focus on measuring and valuing these costs through onset, diagnosis, and post-diagnosis.

Designing, conducting, and reporting reproducible animal experiments.

In biomedicine and many other fields, there are growing concerns around the reproducibility of research findings, with many researchers being unable to replicate their own or others' results. This raises important questions as to the validity and usefulness of much published research. In this review, we aim to engage researchers in the issue of research reproducibility and equip them with the necessary tools to increase the reproducibility of their research. We first highlight the causes and potential impact of non-reproducible research and emphasise the benefits of working reproducibly for the researcher and broader research community. We address specific targets for improvement and steps that individual researchers can take to increase the reproducibility of their work. We next provide recommendations for improving the design and conduct of experiments, focusing on in vivo animal experiments. We describe common sources of poor internal validity of experiments and offer practical guidance for limiting these potential sources of bias at different experimental stages, as well as discussing other important considerations during experimental design. We provide a list of key resources available to researchers to improve experimental design, conduct, and reporting. We then discuss the importance of open research practices such as study preregistration and the use of preprints and describe recommendations around data management and sharing. Our review emphasises the importance of reproducible work and aims to empower every individual researcher to contribute to the reproducibility of research in their field.

Evaluating key performance indicators of the process of care in juvenile idiopathic arthritis.

OBJECTIVE: To determine whether and how often the information to measure a set of key performance indicators (KPIs) in juvenile idiopathic arthritis (JIA) is found in data collected routinely in a Pediatric Rheumatology Clinic. METHODS: A retrospective electronic chart review and administrative data analysis was conducted for a cohort of 140 patients with JIA at a tertiary Pediatric Rheumatology Clinic between 2016-2020. The set of KPIs include measuring patient outcomes (joint assessment, physician's global assessment of disease activity, assessment of functional ability, composite disease activity measurement), access to care (waiting time between referral and first visit, visit with the rheumatologist within the first year of diagnosis, annual follow-up visits with the rheumatologist), and safety (tuberculosis screening, and laboratory monitoring). Documentation was assessed as a binary variable indicating whether the required information was ever found. Documentation frequency for each KPI was assessed with counts and percentages of the number of times the required information was documented for each clinic visit. Compliance with the safety KPI definitions was assessed using administrative databases. RESULTS: Data for each KPI were found at least once in the cohort and documentation varied in frequency and consistency. Access to care and safety KPIs were documented more frequently than patient outcome KPIs. A joint assessment was documented at every visit for 95% of patients, 46% for an assessment of pain, and none for a physician's global assessment of disease activity, an assessment of functional ability, or a composite disease activity measurement. CONCLUSION: Although feasible to measure, there is an opportunity for improving the consistency of documentation. Having an active system of monitoring KPIs and tools to simplify measurement is a key step in the process toward improved patient care outcomes. Streamlining the collection of KPI data can increase the likelihood of compliance. Next steps should involve replicating this study in various centres.

Pharmacological treatment patterns in patients with juvenile idiopathic arthritis in the Netherlands: a real-world data analysis.

OBJECTIVE: To investigate medication prescription patterns among children with JIA, including duration, sequence and reasons for medication discontinuation. METHODS: This study is a single-centre, retrospective analysis of prospective data from the electronic medical records of JIA patients receiving systemic therapy aged 0-18 years between 1 April 2011 and 31 March 2019. Patient characteristics (age, gender, JIA subtype) and medication prescriptions were extracted and analysed using descriptive statistics, Sankey diagrams and Kaplan-Meier survival methods. RESULTS: Over a median of 4.2 years follow-up, the 20 different medicines analysed were prescribed as monotherapy (n = 15) or combination therapy (n = 48 unique combinations) among 236 patients. In non-systemic JIA, synthetic DMARDs were prescribed to almost all patients (99.5%), and always included MTX. In contrast, 43.9% of non-systemic JIA patients received a biologic DMARD (mostly adalimumab or etanercept), ranging from 30.9% for oligoarticular persistent ANA-positive JIA, to 90.9% for polyarticular RF-positive JIA. Among systemic JIA, 91.7% received a biologic DMARD (always including anakinra). When analysing medication prescriptions according to their class, 32.6% involved combination therapy. In 56.8% of patients, subsequent treatment lines were initiated after unsuccessful first-line treatment, resulting in 68 unique sequences. Remission was the most common reason for DMARD discontinuation (44.7%), followed by adverse events (28.9%) and ineffectiveness (22.1%). CONCLUSION: This paper reveals the complexity of pharmacological treatment in JIA, as indicated by: the variety of mono- and combination therapies prescribed, substantial variation in medication prescriptions between subtypes, most patients receiving two or more treatment lines, and the large number of unique treatment sequences.

Transitions from pediatric to adult rheumatology care for juvenile idiopathic arthritis: a patient led qualitative study.

BACKGROUND: Juvenile idiopathic arthritis (JIA) is a childhood autoimmune disease that causes swelling and pain in at least one joint. Young people with JIA experience symptoms that persist into adulthood, and thus will undergo a transition including the o transfer of care from a pediatric rheumatologist an adult rheumatologist. Missing from the literature is research that centres the transition experience of young people with JIA in Canada. This goal of this patient-led research was to explore the experience young people with JIA through the process of transition. METHODS: Qualitative study using the Patient and Community Engaged Research (PaCER) approach. Trained patient-researchers conducted three focus groups using the Set, Collect and Reflect PaCER process. Participants, recruited via purposive and snowball sampling using research/personal networks and social media, were young people with JIA in Canada between 18 and 28 years who had experienced with the process of transition to adult care. Recordings were transcribed verbatim. Patient researchers individually coded overlapping sections of the data, and thematic analysis was conducted. RESULTS: In total, nine individuals participated in one or more focus groups. Three themes were identified, with sub-themes: preparedness for transition (readiness for the transfer of care, developing self-advocacy skills), continuity and breadth of care (changing relationships, culture shock, new responsibilities), need for support (social support, mental health support, and ongoing support needs - beyond the transfer of care. Peer support was a connecting concept in the support sub-themes. Transition was more than a change in primary physician but also a change in the care model and breadth of care provided, which was challenging for young people especially if they had insufficient information. CONCLUSIONS: Transition from pediatric to adult care in rheumatology is a significant period for young people living with JIA, and this patient-led study provided insight into the experience from the perspective of young people with JIA which is critical to informing the development of supports for patients through the process. Patients, caregivers, pediatric and adult rheumatologists and members of the multi-disciplinary care team need to collaborate in terms of resources preparing for transfer, and support throughout the transition process to ensure a successful transition process.

Factors associated with care- and health-related quality of life of caregivers of children with juvenile idiopathic arthritis.

OBJECTIVE: This study investigates the relationship of child, caregiver, and caring context measurements with the care-related quality of life (CRQoL) and health-related quality of life (HRQoL) of caregivers of children with juvenile idiopathic arthritis (JIA). METHODS: We performed a cross-sectional analysis of baseline data on caregivers of children with JIA from Canada and the Netherlands collected for the "Canada-Netherlands Personalized Medicine Network in Childhood Arthritis and Rheumatic Diseases" study from June 2019 to September 2021. We used the CRQoL questionnaire (CarerQoL), adult EQ-5D-5L, and proxy-reported Youth 5-Level version of EuroQoL (EQ-5D-5L-Y) to assess caregiver CRQoL, caregiver HRQoL, and child HRQoL, respectively. We used a multivariate analysis to assess the relationship between both caregiver CRQoL and HRQoL and patient, caregiver, and caring context measurements. RESULTS: A total of 250 caregivers were included in this study. Most of the caregivers were from the Netherlands (n = 178, 71%) and 77% were females (n = 193). The mean CarerQoL scores was 82.7 (standard deviation (SD) 11.4) and the mean EQ-5D-5L utility score was 0.87 (SD 0.16). Child HRQoL and employment had a positive relationship with both caregiver CarerQoL and EQ-5D-5L utility scores (p < 0.05), while receiving paid or unpaid help had a negative relationship with both scores (p < 0.05). CONCLUSION: Our findings indicated that to understand the impact of JIA on families, we need to consider socio-economic factors, such as employment and support to carry caregiving tasks, in addition to child HRQoL.

Effectiveness of biomaterial-based combination strategies for spinal cord repair - a systematic review and meta-analysis of preclinical literature.

STUDY DESIGN: Systematic review and meta-analysis of preclinical literature. OBJECTIVES: To assess the effects of biomaterial-based combination (BMC) strategies for the treatment of Spinal Cord Injury (SCI), the effects of individual biomaterials in the context of BMC strategies, and the factors influencing their efficacy. To assess the effects of different preclinical testing paradigms in BMC strategies. METHODS: We performed a systematic literature search of Embase, Web of Science and PubMed. All controlled preclinical studies describing an in vivo or in vitro model of SCI that tested a biomaterial in combination with at least one other regenerative strategy (cells, drugs, or both) were included. Two review authors conducted the study selection independently, extracted study characteristics independently and assessed study quality using a modified CAMARADES checklist. Effect size measures were combined using random-effects models and heterogeneity was explored using meta-regression with tau2, I2 and R2 statistics. We tested for small-study effects using funnel plot-based methods. RESULTS: 134 publications were included, testing over 100 different BMC strategies. Overall, treatment with BMC therapies improved locomotor recovery by 25.3% (95% CI, 20.3-30.3; n = 102) and in vivo axonal regeneration by 1.6 SD (95% CI 1.2-2 SD; n = 117) in comparison with injury only controls. CONCLUSION: BMC strategies improve locomotor outcomes after experimental SCI. Our comprehensive study highlights gaps in current knowledge and provides a foundation for the design of future experiments.

Real-world data reveals the complexity of disease modifying anti-rheumatic drug treatment patterns in juvenile idiopathic arthritis: an observational study.

OBJECTIVE: Pharmacological treatment is a cornerstone of care for children with juvenile idiopathic arthritis (JIA). The objective of this study is to evaluate prescription patterns of conventional and biologic disease modifying anti-rheumatic drugs (c-DMARDs and b-DMARDs) for patients with JIA. METHODS: We conducted a retrospective cohort study of children diagnosed with JIA at a rheumatology pediatric clinic. Eligibility criteria were defined as children and youth newly diagnosed with enthesis-related arthritis, polyarticular, or oligoarticular JIA between 2011 and 2019, with at least one year of observation. Data on c-DMARDs and b-DMARDs prescriptions were obtained from electronic medical charts. We used descriptive statistics, Kaplan-Meier survival methods, and Sankey diagrams to describe treatment prescription patterns. RESULTS: A total of 325 patients with JIA were included, with a median observation time of 3.7 years. The most frequently prescribed c-DMARD and b-DMARD were methotrexate and etanercept, respectively. Within the first year of rheumatology care, 62% and 21% of patients had a c-DMARD and a b-DMARD prescribed, respectively. These proportions varied greatly by JIA subtype. Among the 147 (147/325, 45%) patients that had at least one b-DMARD prescribed, 24% were prescribed a second, and 7% a third-line of b-DMARD. A total of 112 unique treatment sequences were observed, with c-DMARD monotherapy followed by the addition of either a b-DMARD (56%) or another c-DMARD (30%) being the two most prevalent patterns in this cohort. CONCLUSION: We observed a variety of treatment trajectories, with many patients experiencing multiple treatment lines, illustrating the complexity of the overall JIA treatment path.

Factors That Patients Consider in Their Choice of Non-Surgical Management for Hip and Knee Osteoarthritis: Formative Qualitative Research for a Discrete Choice Experiment.

BACKGROUND: Approximately half of patients with hip and knee osteoarthritis have tried non-surgical management before surgical consultation. Understanding the many factors affecting the uptake of recommended strategies is important to inform future development of such management strategies. OBJECTIVES: The aim of this study was to explore and identify factors that patients with osteoarthritis consider when choosing non-surgical management for hip and knee osteoarthritis, as formative research for a study of patient preferences for non-surgical management programs for osteoarthritis. METHODS: A qualitative research design was used. Participants were recruited using a combination of stratified and convenience sampling. Interviews were conducted, using a semi-structured interview guide, with English-speaking patients who had self-reported hip and/or knee osteoarthritis and at least one joint that had not undergone replacement surgery. Data were thematically analyzed. RESULTS: Thirteen patients participated in these interviews. Sixteen factors that participants considered when choosing non-surgical osteoarthritis management were identified. Eleven were extrinsic, relating to features of programs and services, and are categorized as types of interventions, general program and service details, and program-specific details. Five were intrinsic to the individual and influenced how decisions for osteoarthritis management were approached and the options available to choose from. Three novel factors included participants' desire for further management, their views about joint replacement surgery, and whether they felt personal choice was available in osteoarthritis management strategies. CONCLUSION: Key factors were identified that patients considered when making decisions about non-surgical management for their osteoarthritis that will be used to inform a discrete choice experiment (DCE) that aims to measure preferences for these factors.

Perspectives of Pediatric Rheumatologists on Initiating and Tapering Biologics in Patients with Juvenile Idiopathic Arthritis: A Formative Qualitative Study.

BACKGROUND: Few studies have examined pediatric rheumatologists' approaches to treatment decision making for biologic therapy for patients with juvenile idiopathic arthritis (JIA). This study presents the qualitative research undertaken to support the development of a Best-Worst Scaling (BWS) survey for tapering in JIA. The study objectives were to (1) describe the treatment decision-making process of pediatric rheumatologists to initiate and taper biologics; and (2) select attributes for a BWS survey. METHODS: Pediatric rheumatologists across Canada were recruited to participate in interviews using purposeful sampling. Interviews were conducted until saturation was achieved. Interview recordings were transcribed verbatim and transcripts were analyzed using deductive thematic analysis. Initial codes were organized into themes and subthemes using an iterative process. Attributes for the BWS survey were developed from these themes and a literature review was conducted in parallel to inform survey development. Further refinement of the attributes was done through consultation with the research team. RESULTS: Five pediatric rheumatologists participated in the interviews. Shared decision making was part of the approach to initiating and tapering biologics in their practice. Tapering approaches differed; some pediatric rheumatologists preferred to stop biologics immediately, while others tapered by reducing dose and/or increasing the dose interval over time. A total of 14 attributes were developed for the BWS. Thirteen attributes were selected from the themes that emerged from the qualitative interviews and one attribute was included after review with the research team. Attributes related to patient characteristics included JIA subtype, time in remission, history or presence of joint damage or erosive disease, how challenging it was to achieve remission, and history of flares. Contextual attributes included accessibility of biologics and willingness to taper biologics. CONCLUSION: This study contributes to the limited literature on pediatric rheumatologists' approaches to treatment decision making for biologics in JIA and identifies attributes that affect the decision to both initiate and taper. Further research is planned to implement the BWS survey to understand the importance of the attributes identified. Additional investigation is required to determine if these characteristics align with patient and parent preferences.

Patient beliefs about who and what influences their hip and knee osteoarthritis symptoms and progression.

BACKGROUND: Osteoarthritis management aims to reduce pain and improve function. Many factors affect whether patients follow recommended strategies. Locus of control refers to individual beliefs around who and what influences health. Locus of control is related to the treatment strategies patients prefer. Currently, no studies explore locus of control in non-surgical management of osteoarthritis. OBJECTIVES: To explore patients' beliefs about the influences on their osteoarthritis symptoms and disease progression. METHODS: Semi-structured interviews were conducted with individuals experiencing self-reported hip and/or knee osteoarthritis who had at least one joint that had not undergone replacement surgery. We used a qualitative description approach and the Braun and Clarke method for thematic analysis. Participants' locus of control classifications-internal, chance, doctors, or other people-were based on the Multidimensional Health Locus of Control (MHLC) Scales Form C score. RESULTS: Locus of control was discussed in relation to aetiology, progression, and symptoms. Participants' opinions varied on whether their osteoarthritis progression could be influenced. 46% of participants attributed control to other people. Most participants believed that a previous injury had caused their osteoarthritis and that both themselves and others had some influence over their osteoarthritis symptoms, regardless of their locus of control classification. CONCLUSION: This research highlights the need for education about: the aetiology of osteoarthritis, the link between management and progression, and patient management of osteoarthritis. Further research is required to discern why expected patterns were not observed between participants' beliefs and locus of control classifications.

An Economic Evaluation of Disallowing Body Checking in 11- to 12-Year-Old Ice Hockey Leagues.

BACKGROUND: After a national policy change in 2013 disallowing body checking in Pee Wee ice hockey games, the rate of injury was reduced by 50% in Alberta. However, the effect on associated health care costs has not been examined previously. HYPOTHESIS: A national policy removing body checking in Pee Wee (ages 11-12 years) ice hockey games will reduce injury rates, as well as costs. STUDY DESIGN: Cost-effectiveness analysis alongside cohort study. LEVEL OF EVIDENCE: Level 3. METHODS: A cost-effectiveness analysis was conducted alongside a cohort study comparing rates of game injuries in Pee Wee hockey games in Alberta in a season when body checking was allowed (2011-2012) with a season when it was disallowed after a national policy change (2013-2014). The effectiveness measure was the rate of game injuries per 1000 player-hours. Costs were estimated based on associated health care use from both the publicly funded health care system and privately paid health care cost perspectives. Probabilistic sensitivity analysis was conducted using bootstrapping. RESULTS: Disallowing body checking significantly reduced the rate of game injuries (-2.21; 95% CI [-3.12, -1.31] injuries per 1000 player-hours). We found no statistically significant difference in public health care system (-$83; 95% CI [-$386, $220]) or private health care costs (-$70; 95% CI [-$198, $57]) per 1000 player-hours. The probability that the policy of disallowing body checking was dominant (with both fewer injuries and lower costs) from the perspective of the public health care system and privately paid health care was 78% and 92%, respectively. CONCLUSION: Given the significant reduction in injuries, combined with lower public health care system and private costs in the large majority of iterations in the probabilistic sensitivity analysis, our findings support the policy change disallowing body checking in ice hockey in 11- and 12-year-old ice hockey leagues.

Costs of Hospital-Associated Care for Patients With Juvenile Idiopathic Arthritis in the Dutch Health Care System.

OBJECTIVE: The aim of this study was to quantify costs of hospital-associated care for juvenile idiopathic arthritis (JIA), provide insights in patient-level variation in costs, and investigate costs over time from the moment of JIA diagnosis. Results were reported for all JIA patients in general and by subtype. METHODS: This study was a single-center, retrospective analysis of prospective data from electronic medical records of children with JIA, ages 0-18 years, between April 1, 2011 and March 31, 2019. Patient characteristics (age, sex, JIA subtype) and hospital-based resource use (consultations, medication, radiology procedures, laboratory testing, surgeries, emergency department [ED] visits, hospital stays) were extracted and analyzed. Unit prices were obtained from Dutch reimbursement lists and pharmaceutical and hospital list prices. RESULTS: The analysis included 691 patients. The mean total cost of hospital care was €3,784/patient/year, of which €2,103 (55.6%) was attributable to medication. Other costs involved pediatric rheumatologist visits (€633/patient/year [16.7%]), hospital stays (€439/patient/year [11.6%]), other within-hospital specialist visits (€324/patient/year [8.6%]), radiology procedures (€119/patient/year [3.1%]), laboratory tests (€114/patient/year [3.0%]), surgeries (€46/patient/year [1.2%]), and ED visits (€6/patient/year [0.2%]). Mean annual total costs varied between JIA subtypes and between individuals and were the highest for systemic JIA (€7,772/patient/year). Over the treatment course, costs were the highest in the first month after JIA diagnosis. CONCLUSION: Hospital care costs of JIA vary substantially between individuals, between subtypes, and over the treatment course. The highest annual costs were for systemic JIA, primarily attributable to medication (i.e., biologics). Costs of other hospital-associated care were comparable regardless of subtype.

Reality Check 2: The Cost-Effectiveness of Policy Disallowing Body Checking in Non-Elite 13- to 14-Year-Old Ice Hockey Players.

Sport-related injuries are the leading cause of injury in youth and are costly to the healthcare system. When body checking is disallowed in non-elite levels of Bantam (ages 13-14 years) ice hockey, the injury rate is reduced, but the impact on costs is unknown. This study compared rates of game injuries and costs among non-elite Bantam ice hockey leagues that disallow body checking to those that did not. Methods: An economic evaluation was conducted alongside a prospective cohort study comparing 608 players from leagues where body checking was allowed in games (Calgary/Edmonton 2014-2015, Edmonton 2015-2016) with 396 players from leagues where it was not allowed in games (Vancouver, Kelowna 2014-2015, Calgary in 2015-2016). The effectiveness measure was rate of game injuries per 1000 player-hours. Costs were estimated based on associated healthcare use within the publicly funded healthcare system as well as privately paid healthcare costs. Probabilistic sensitivity analysis was conducted using bootstrapping. Results: Disallowing body checking reduced the rate of injuries by 4.32 per 1000 player-hours (95% CI -6.92, -1.56) and reduced public and total healthcare system costs by $1556 (95% CI -$2478, -$559) and $1577 (95% CI -$2629, -$500) per 1000 player-hours, respectively. These finding were robust in over 99% of iterations in sensitivity analyses in the public healthcare and the total healthcare system perspectives. There was no statistically significant difference in privately paid healthcare costs (-$65 per 1000 player-hours (95% CI -$220, $99)). Interpretation: Disallowing body checking in non-elite 13-14-year-old ice hockey nationally would prevent injuries and reduce public healthcare costs.