Abstract
SummaryBackgroundJuvenile idiopathic arthritis (JIA) is a heterogeneous disease, the signs and symptoms of which can be summarised with use of composite disease activity measures, including the clinical Juvenile Arthritis Disease Activity Score (cJADAS). However, clusters of children and young people might experience different global patterns in their signs and symptoms of disease, which might run in parallel or diverge over time. We aimed to identify such clusters in the 3 years after a diagnosis of JIA. The identification of these clusters would allow for a greater understanding of disease progression in JIA, including how physician-reported and patient-reported outcomes relate to each other over the JIA disease course.MethodsIn this multicentre prospective longitudinal study, we included children and young people recruited before Jan 1, 2015, to the Childhood Arthritis Prospective Study (CAPS), a UK multicentre inception cohort. Participants without a cJADAS score were excluded. To assess groups of children and young people with similar disease patterns in active joint count, physician's global assessment, and patient or parental global evaluation, we used latent profile analysis at initial presentation to paediatric rheumatology and multivariate group-based trajectory models for the following 3 years. Optimal models were selected on the basis of a combination of model fit, clinical plausibility, and model parsimony.FindingBetween Jan 1, 2001, and Dec 31, 2014, 1423 children and young people with JIA were recruited to CAPS, 239 of whom were excluded, resulting in a final study population of 1184 children and young people. We identified five clusters at baseline and six trajectory groups using longitudinal follow-up data. Disease course was not well predicted from clusters at baseline; however, in both cross-sectional and longitudinal analyses, substantial proportions of children and young people had high patient or parent global scores despite low or improving joint counts and physician global scores. Participants in these groups were older, and a higher proportion of them had enthesitis-related JIA and lower socioeconomic status, compared with those in other groups.InterpretationAlmost one in four children and young people with JIA in our study reported persistent, high patient or parent global scores despite having low or improving active joint counts and physician's global scores. Distinct patient subgroups defined by disease manifestation or trajectories of progression could help to better personalise health-care services and treatment plans for individuals with JIA.FundingMedical Research Council, Versus Arthritis, Great Ormond Street Hospital Children's Charity, Olivia's Vision, and National Institute for Health Research.
Highlights
Juvenile idiopathic arthritis (JIA) is a heterogeneous condition with onset in childhood or early adolescence and common disease features that include joint swelling and pain.[1]
Between Jan 1, 2001, and Dec 31, 2014, 1423 children and young people with JIA were recruited to Childhood Arthritis Prospective Study (CAPS), 239 of whom were excluded, resulting in a final study population of 1184 children and young people
Our study shows that novel unsupervised machine learning methods applied to traditional epidemiological data represent an exciting step toward stratified management for children and young people with JIA, as new avenues for biomarker discovery and further understanding of disease mechanisms become available within this heterogeneous disease
Summary
Juvenile idiopathic arthritis (JIA) is a heterogeneous condition with onset in childhood or early adolescence and common disease features that include joint swelling and pain.[1]. Key outcomes for individuals with JIA are included in a core outcome set and are incorporated into the juve nile arthritis disease activity score (JADAS) and clinical. JADAS (cJADAS),[4,5] two composite outcome measures of JIA. JADAS allows for a single measure of disease activity in individuals with JIA, the individual components of the score do not always correlate.[6,7] This is evident for components measured by the physician (active joint count or the physician’s global assessment) versus those reported by the patient (such as parent or patient global evaluation). A quarter of children and young people (aged younger than 16 years) with clinically inactive disease (a disease state defined by clinician-only measured markers of disease activity) have ongoing symptoms,[8] including disability, in Lancet Rheumatol 2021; 3: e111–21
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