Development and external validation of a model predicting new-onset chronic uveitis at different disease durations in juvenile idiopathic arthritis
Paediatric Rheumatology International Trials Organisation (PRINTO), UK CAPS study and German ICON study
(2023) Arthritis & Rheumatology, volume 75, issue 2, pp. 318 - 327
(Article)
Abstract
Objective: To develop and externally validate a prediction model for new-onset chronic uveitis in children with juvenile idiopathic arthritis (JIA) for clinical application. Methods: Data from the international Pharmachild registry were used to develop a multivariable Cox proportional hazards model. Predictors were selected by backward selection, and missing values were
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handled by multiple imputation. The model was subsequently validated and recalibrated in 2 inception cohorts: the UK Childhood Arthritis Prospective Study (CAPS) study and the German Inception Cohort of Newly diagnosed patients with juvenile idiopathic arthritis (ICON) study. Model performance was evaluated by calibration plots and C statistics for the 2-, 4-, and 7-year risk of uveitis. A diagram and digital risk calculator were created for use in clinical practice. Results: A total of 5,393 patients were included for model development, and predictor variables were age at JIA onset (hazard ratio [HR] 0.83 [95% confidence interval (95% CI) 0.77–0.89]), ANA positivity (HR 1.59 [95% CI 1.06–2.38]), and International League of Associations for Rheumatology category of JIA (HR for oligoarthritis, psoriatic arthritis, and undifferentiated arthritis versus rheumatoid factor–negative polyarthritis 1.40 [95% CI 0.91–2.16]). Performance of the recalibrated prediction model in the validation cohorts was acceptable; calibration plots indicated good calibration and C statistics for the 7-year risk of uveitis (0.75 [95% CI 0.72–0.79]) for the ICON cohort and 0.70 [95% CI 0.64–0.76] for the CAPS cohort). Conclusion: We present for the first time a validated prognostic tool for easily predicting chronic uveitis risk for individual JIA patients using common clinical parameters. This model could be used by clinicians to inform patients/parents and provide guidance in choice of uveitis screening frequency and arthritis drug therapy.
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Keywords: Immunology and Allergy, Rheumatology, Immunology, Journal Article
ISSN: 2326-5191
Publisher: John Wiley and Sons Ltd
Note: Funding Information: The authors thank all PRINTO centers for their contribution to the data collection and PRINTO research assistants (Chiara Pallotti, Silvia Scala, Simona Angioloni, and Luca Villa), and we acknowledge ERN-RITA for provided expertise. We also thank all principal investigators, clinical staff, and research coordinators of CAPS who have made this research possible, as well as members of the research team at the University of Manchester, UK. These include: G. Cleary, E. Baildam (Alder Hey Children's Hospital, UK), L. Wedderburn (Great Ormond Street Hospital, UK), J. Davidson (Royal Hospital for Sick Children, Edinburgh and Royal Hospital for Children, Glasgow, UK), A. Chieng (Royal Manchester Children's Hospital, UK), F. McErlane, H. Foster (Royal Victoria Infirmary, UK), C. Ciurtin, Y. Ioannou (University College London Hospital, UK) and W. Thomson, K. Hyrich (University of Manchester). We would also like to acknowledge the contributions of the following ICON JIA Study Group collaborators: Tilmann Kallinich (Universitätsmedizin Charité Berlin), Frank Dressler (Medizinische Hochschule Hannover), Jasmin Kümmerle Deschner (Universität Tübingen), Frank Weller-Heinemann (Prof. Hess Kinderklinik Bremen), Gerd Horneff (Asklepios Kinderklinik Sankt Augustin), Anton Hospach (Olgahospital Stuttgart), Kirsten Mönkemöller (Kinderkrankenhaus der Stadt Köln), Johannes Peter Haas (Deutsches Zentrum für Kinder- und Jugendrheumatologie Garmisch Partenkirchen), Daniel Windschall (St. Joseph Stift Sendenhorst), Ivan Foeldvari (Kinderrheumatologische Praxis am AK Eilbek Hamburg), Dirk Foell (Klinik für Pädiatrische Rheumatologie und Immunologie, Universitätsklinikum Münster). Funding Information: Supported by FOREUM Foundation for Research in Rheumatology. Pharmachild was supported by the European Union (grant 260353) and by funding from the Italian public hospital IRCCS Istituto Giannina Gaslini. CAPS was supported by Versus Arthritis (UK grant 20542). The research team at The University of Manchester were supported by the Centre for Epidemiology Versus Arthritis (UK grant 21755), the Centre for Genetics and Genomics Versus Arthritis (UK grant 21754), and the NIHR Manchester Biomedical Research Centre. The ICON study was supported by the German Federal Ministry of Education and Research (awards BMBF, FKZ 01ER0812, 01ER0813, 01ER0828, FKZ‐01‐ER1504A, 01‐ER‐1504B, and 01‐ER‐1504C). Publisher Copyright: © 2022 The Authors. Arthritis & Rheumatology published by Wiley Periodicals LLC on behalf of American College of Rheumatology.
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