Abstract
Objective
To assess long-term outcomes of children with JIA diagnosed in the biologic era.
Methods
Chart review of patients prospectively enrolled in the Research in Arthritis in Canadian ...Children Emphasizing Outcomes inception cohort at two Canadian centres. Inactive disease and remission were defined according to Wallace criteria.
Results
We included 247 of 254 (97%) eligible patients diagnosed 2005–10. At the last follow-up visit at a median age of 16.9 years, 47% were in remission off medications, 25% in remission on medications and 27% had active disease; 51% were on at least one anti-rheumatic medication (22% on biologics). Patients with systemic JIA had the highest frequency of remission off medications (70%) and patients with RF-positive polyarthritis had the lowest (18%) (P <0.05 by Fisher’s exact test). Among 99 patients with oligoarthritis at enrolment, 14 (14%) had an oligoarthritis extended course. Forty-five patients (18%) had at least one erosion or joint space narrowing in X-rays or MRI, and two (0.8%) required joint replacement.
Conclusion
Relative to historical cohorts, this study suggests a reduction in JIA permanent damage, a more favourable prognosis for systemic JIA and a lower progression to oligoarthritis extended category. However, in an era of biologic therapy, one in four patients with JIA still enter adulthood with active disease and one in two still on treatment.
Young adults living with episodic disabilities face unpredictable disruptions to their employment and health. Our study aimed to examine the impact of employment and income support interventions on ...the health and well-being of young adults living with episodic disabilities.
We conducted a systematic review of peer-reviewed intervention studies published in 2001-2021 in industrialized contexts. Two independent reviewers screened titles, abstracts and full-texts. We undertook a narrative synthesis of eligible articles.
Our search yielded 15 269 published articles, of which only five studies were eligible for evidence synthesis. All articles were appraised as being of medium quality. Four interventions focused on young adults living with mental health conditions. Two were based in clinical settings; three were based in community-based settings. Each employment intervention exhibited improvements in health outcomes. Three studies examined the impact of supported employment interventions that were particularly beneficial to improving work and health outcomes.
Involvement in employment interventions could provide benefits for the health of young adults living with episodic disabilities. Our systematic review highlights the need for research to elaborate on the ways in which employment interventions can impact the health and well-being of young adults living with different episodic disabilities.
Objective
The COVID‐19 pandemic has had considerable economic repercussions for young workers. The current study was undertaken to examine the impact of the pandemic on the employment of young adults ...with rheumatic disease and on perceptions of work and health.
Methods
Surveys were administered to young adults with rheumatic disease prior to and following the onset of the COVID‐19 pandemic. Surveys asked about employment status and collected information on sociodemographic, disease/health, and work‐context factors. Items also asked about the perceived impact of the COVID‐19 pandemic on work and health. A generalized estimating equation model was fitted to examine the effect of the pandemic on employment.
Results
In total, 133 young adults completed the pre–COVID‐19 pandemic survey (mean age 28.9 years, 82% women). When compared to the pre–COVID‐19 pandemic period, employment decreased from 86% to 71% following the pandemic, but no other changes were identified in sociodemographic, disease/health, or work‐context factors. The time period following the COVID‐19 pandemic was associated with a 72% lower odds of employment compared to the pre‐pandemic period (odds ratio 0.28 95% confidence interval 0.11–0.71). Those with a postsecondary education or who reported more mental job demands were more likely to be employed following the onset of the pandemic. Also, a majority of participants reported that the pandemic affected health care (83%), treatment access (54%), working conditions (92%), and occupational health and safety (74%).
Conclusion
The onset of the COVID‐19 pandemic had socioeconomic implications for young people with rheumatic disease. To support economic recovery for individuals with rheumatic disease, strategies to promote employment should be designed that account for the young adult life phase and occupational characteristics.
To describe clinical outcomes of juvenile idiopathic arthritis (JIA) in a prospective inception cohort of children managed with contemporary treatments.
Children newly diagnosed with JIA at 16 ...Canadian paediatric rheumatology centres from 2005 to 2010 were included. Kaplan-Meier survival curves for each JIA category were used to estimate probability of ever attaining an active joint count of 0, inactive disease (no active joints, no extraarticular manifestations and a physician global assessment of disease activity <10 mm), disease remission (inactive disease >12 months after discontinuing treatment) and of receiving specific treatments.
In a cohort of 1104 children, the probabilities of attaining an active joint count of 0 exceeded 78% within 2 years in all JIA categories. The probability of attaining inactive disease exceeded 70% within 2 years in all categories, except for RF-positive polyarthritis (48%). The probability of discontinuing treatment at least once was 67% within 5 years. The probability of attaining remission within 5 years was 46-57% across JIA categories except for polyarthritis (0% RF-positive, 14% RF-negative). Initial treatment included joint injections and non-steroidal anti-inflammatory drugs for oligoarthritis, disease-modifying antirheumatic drugs (DMARDs) for polyarthritis and systemic corticosteroids for systemic JIA.
Most children with JIA managed with contemporary treatments attain inactive disease within 2 years of diagnosis and many are able to discontinue treatment. The probability of attaining remission within 5 years of diagnosis is about 50%, except for children with polyarthritis.
We studied an inception cohort of children with juvenile idiopathic arthritis (JIA) to (1) identify distinct disease courses based on changes over 5 years in 5 variables prioritized by patients, ...parents, and clinicians; and (2) estimate the probability of a severe disease course for each child at diagnosis.
Assessments of quality of life, pain, medication requirements, patient-reported side effects, and active joint counts were scheduled at 0, 6, 12, 18, 24, 36, 48, and 60 months. Patients who attended at least 6 assessments were included. Multivariable cluster analysis, r
, and silhouette statistics were used to identify distinct disease courses. One hundred candidate prediction models were developed in random samples of 75% of the cohort; their reliability and accuracy were tested in the 25% not used in their development.
Four distinct courses were identified in 609 subjects. They differed in prioritized variables, disability scores, and probabilities of attaining inactive disease and remission. We named them Mild (43.8% of children), Moderate (35.6%), Severe Controlled (9%), and Severe Persisting (11.5%). A logistic regression model using JIA category, active joint count, and pattern of joint involvement at enrollment best predicted a severe disease course (Controlled + Persisting, c-index = 0.87); 91% of children in the highest decile of risk actually experienced a severe disease course, compared to 5% of those in the lowest decile.
Children in this JIA cohort followed 1 of 4 disease courses and the probability of a severe disease course could be estimated with information available at diagnosis.
Abstract
Objectives
To evaluate the feasibility and preliminary effectiveness of iCanCope with Pain (iCanCope), a smartphone-based pain self-management program, in adolescents with JIA. iCanCope ...featured symptom tracking, goal-setting, pain coping skills and social support.
Methods
A two-arm pilot randomized controlled trial was used to evaluate the iCanCope app compared with a version with symptom tracking only. Primary (feasibility) outcomes were: participant accrual/attrition rates, success of app deployment, acceptability and adherence. Secondary (preliminary effectiveness) outcomes were: pain intensity, pain-related activity limitations and health-related quality of life. Outcomes were assessed at baseline and 8 weeks. Adherence was defined as the proportion of completed symptom reports: ‘low’ (≤24%); ‘low-moderate’ (25–49%); ‘high-moderate’ (50–75%); or ‘high’ (76–100%). Linear mixed models were applied for preliminary effectiveness analyses as per intention-to-treat.
Results
Adolescents (N = 60) were recruited from three paediatric rheumatology centres. Rates of accrual and attrition were 82 and 13%, respectively. Both apps were deployed with high success (over 85%) and were rated as highly acceptable. Adherence was similar for both groups, with most participants demonstrating moderate-to-high adherence. Both groups exhibited a clinically meaningful reduction in pain intensity (≥1 point) that did not statistically differ between groups. There were no significant changes in activity limitations or health-related quality of life.
Conclusion
The iCanCope pilot randomized controlled trial was feasible to implement in a paediatric rheumatology setting. Both apps were deployed successfully, with high acceptability, and were associated with moderate-to-high adherence. Preliminary reductions in pain intensity warrant a future trial to evaluate effectiveness of iCanCope in improving health outcomes in adolescents with JIA.
Trial registration
ClinicalTrials.gov identifier: NCT02764346.
Introduction
The future of work is characterized by changes that could disrupt all aspects of the nature and availability of work. Our study aims to understand how the future of work could result in ...conditions, which contribute to vulnerability for different groups of workers.
Methods
A horizon scan was conducted to systematically identify and synthesize diverse sources of evidence, including academic and gray literature and resources shared over social media. Evidence was synthesized, and trend categories were developed through iterative discussions among the research team.
Results
Nine trend categories were uncovered, which included the digital transformation of the economy, artificial intelligence (AI)/machine learning‐enhanced automation, AI‐enabled human resource management systems, skill requirements for the future of work; globalization 4.0, climate change and the green economy, Gen Zs and the work environment; populism and the future of work, and external shocks to accelerate the changing nature of work. The scan highlighted that some groups of workers may be more likely to experience conditions that contribute to vulnerability, including greater exposure to job displacement or wage depression. The future of work could also create opportunities for labor market engagement.
Conclusion
The future of work represents an emerging public health concern. Exclusion from the future of work has the potential to widen existing social and health inequities. Thus, tailored supports that are resilient to changes in the nature and availability of work are required for workers facing vulnerability.
Objective
The aim of the Paediatric Rheumatology International Trials Organisation (PRINTO) juvenile idiopathic arthritis (JIA) classification criteria, which is still in development, is to identify ...homogeneous groups of JIA patients. This study was undertaken to compare International League of Associations for Rheumatology (ILAR) JIA classification criteria and PRINTO JIA classification criteria using data from the ReACCh‐Out (Research in Arthritis in Canadian Children, Emphasizing Outcomes) cohort.
Methods
We used clinicobiologic data recorded within 7 months of diagnosis to assign a diagnosis of JIA and identify subcategories of JIA among 1,228 patients according to the 2 JIA classification systems. We compared the proportions of patients classified and the alignment of classification categories with clinicobiologic subtypes and adult arthritis types.
Results
The PRINTO criteria classified 244 patients (19.9%) as having early‐onset antinuclear antibody–positive JIA, 157 (12.8%) as having enthesitis/spondylitis–related JIA, 38 (3.1%) as having systemic JIA, and 10 (0.8%) as having rheumatoid factor–positive JIA. A total of 12% of patients were unclassifiable using the ILAR criteria, while 63.3% were unclassifiable using the PRINTO criteria (777 with other JIA and 2 with unclassified JIA). In sensitivity analyses, >50% of patients remained unclassifiable using the PRINTO criteria. Compared to the PRINTO criteria, ILAR JIA categories aligned better with clinicobiologic subtypes in 131 patients (χ2 = 44, P = 0.005, versus χ2 = 15, P = 0.07 for PRINTO), and ILAR categories aligned better with adult types of arthritis in 389 evaluable patients.
Conclusion
Currently identified PRINTO disorders can only be used to classify a minority of JIA patients, leaving a large proportion of JIA patients with other disorders requiring further characterization. Current PRINTO JIA classification criteria do not align better with clinicobiologic subtypes or adult forms of arthritis compared with the older ILAR classification system.
Abstract Objectives Clinicians need to be aware of the growing list of defined monogenic etiologies of autoimmune diseases. This is particularly relevant when evaluating children, as these rare ...monogenic forms of autoimmunity tend to present very early in life. Methods and results By harnessing the transformative power of next generation sequencing, we made the unifying diagnosis of RAS-associated autoimmune leukoproliferative disease (RALD), caused by the somatic gain-of-function p.G13C KRAS mutation, in a boy with the seemingly unrelated immune dysregulatory conditions of Rosai-Dorfman and systemic lupus erythematosis (SLE). Conclusions This case expands our understanding of the clinical phenotypes associated with the extremely rare condition of RALD, and emphasizes the importance of always considering the possibility of a monogenic cause for autoimmunity, particularly when the disease manifestations begin early in life and do not follow a typical clinical course.
Abstract
Objective
To identify differences between baseline Canadian JIA practices and the 2019 ACR guidelines for JIA.
Methods
Canadian paediatric rheumatologists were surveyed for their opinions on ...reasonable a priori target adherence rates for JIA guideline recommendations. Prospectively collected data for 266 newly diagnosed children from 2017 to 2019 were analysed to calculate observed adherence rates. Kaplan–Meier survival curves were used to estimate the cumulative incidence of starting synthetic or biologic DMARDs (sDMARD or bDMARD, respectively) for different patient groups.
Results
A total of 25/61 (41%) eligible physicians answered the survey. Most survey respondents (64%) felt that adherence targets should vary depending on the strength of the recommendation and quality of evidence, from a mean of 84% for strong recommendations with high-quality evidence to 29% for conditional recommendations with very low-quality evidence. Data showed 13/19 (68%) recommendations would have met proposed targets and 10/19 (53%) had ≥80% observed adherence. Exceptions were the use of subcutaneous vs oral MTX (53%) and infrequent treatment escalation from NSAIDs to bDMARDs in patients with sacroiliitis (31%) or enthesitis (0%). By 12 weeks, 95% of patients with polyarthritis received sDMARDs, 38% of patients with systemic JIA received bDMARDs and 22% of patients with sacroiliitis received bDMARDs.
Conclusion
Canadian paediatric rheumatology practices were in line with many 2019 JIA guideline recommendations before their publication, except for frequent use of oral MTX and infrequent direct escalation from NSAIDs to bDMARDs in sacroiliitis and enthesitis.
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