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Development and External Validation of a Model Predicting New‐Onset Chronic Uveitis at Different Disease Durations in Juvenile Idiopathic Arthritis

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....

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Autores principales: van Straalen, Joeri W., Kearsley‐Fleet, Lianne, Klotsche, Jens, de Roock, Sytze, Minden, Kirsten, Heiligenhaus, Arnd, Hyrich, Kimme L., de Boer, Joke H., Lamot, Lovro, Olivieri, Alma N., Gallizzi, Romina, Smolewska, Elzbieta, Faugier, Enrique, Pastore, Serena, Hashkes, Philip J., Herrera, Cristina N., Emminger, Wolfgang, Consolini, Rita, Wulffraat, Nico M., Ruperto, Nicolino, Swart, Joost F.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Wiley Periodicals, Inc. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10108055/
https://www.ncbi.nlm.nih.gov/pubmed/36054539
http://dx.doi.org/10.1002/art.42329
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author van Straalen, Joeri W.
Kearsley‐Fleet, Lianne
Klotsche, Jens
de Roock, Sytze
Minden, Kirsten
Heiligenhaus, Arnd
Hyrich, Kimme L.
de Boer, Joke H.
Lamot, Lovro
Olivieri, Alma N.
Gallizzi, Romina
Smolewska, Elzbieta
Faugier, Enrique
Pastore, Serena
Hashkes, Philip J.
Herrera, Cristina N.
Emminger, Wolfgang
Consolini, Rita
Wulffraat, Nico M.
Ruperto, Nicolino
Swart, Joost F.
author_facet van Straalen, Joeri W.
Kearsley‐Fleet, Lianne
Klotsche, Jens
de Roock, Sytze
Minden, Kirsten
Heiligenhaus, Arnd
Hyrich, Kimme L.
de Boer, Joke H.
Lamot, Lovro
Olivieri, Alma N.
Gallizzi, Romina
Smolewska, Elzbieta
Faugier, Enrique
Pastore, Serena
Hashkes, Philip J.
Herrera, Cristina N.
Emminger, Wolfgang
Consolini, Rita
Wulffraat, Nico M.
Ruperto, Nicolino
Swart, Joost F.
author_sort van Straalen, Joeri W.
collection PubMed
description 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 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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spelling pubmed-101080552023-04-18 Development and External Validation of a Model Predicting New‐Onset Chronic Uveitis at Different Disease Durations in Juvenile Idiopathic Arthritis van Straalen, Joeri W. Kearsley‐Fleet, Lianne Klotsche, Jens de Roock, Sytze Minden, Kirsten Heiligenhaus, Arnd Hyrich, Kimme L. de Boer, Joke H. Lamot, Lovro Olivieri, Alma N. Gallizzi, Romina Smolewska, Elzbieta Faugier, Enrique Pastore, Serena Hashkes, Philip J. Herrera, Cristina N. Emminger, Wolfgang Consolini, Rita Wulffraat, Nico M. Ruperto, Nicolino Swart, Joost F. Arthritis Rheumatol Pediatric Rheumatology 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 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. Wiley Periodicals, Inc. 2022-12-13 2023-02 /pmc/articles/PMC10108055/ /pubmed/36054539 http://dx.doi.org/10.1002/art.42329 Text en © 2022 The Authors. Arthritis & Rheumatology published by Wiley Periodicals LLC on behalf of American College of Rheumatology. https://creativecommons.org/licenses/by/4.0/This is an open access article under the terms of the http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited.
spellingShingle Pediatric Rheumatology
van Straalen, Joeri W.
Kearsley‐Fleet, Lianne
Klotsche, Jens
de Roock, Sytze
Minden, Kirsten
Heiligenhaus, Arnd
Hyrich, Kimme L.
de Boer, Joke H.
Lamot, Lovro
Olivieri, Alma N.
Gallizzi, Romina
Smolewska, Elzbieta
Faugier, Enrique
Pastore, Serena
Hashkes, Philip J.
Herrera, Cristina N.
Emminger, Wolfgang
Consolini, Rita
Wulffraat, Nico M.
Ruperto, Nicolino
Swart, Joost F.
Development and External Validation of a Model Predicting New‐Onset Chronic Uveitis at Different Disease Durations in Juvenile Idiopathic Arthritis
title Development and External Validation of a Model Predicting New‐Onset Chronic Uveitis at Different Disease Durations in Juvenile Idiopathic Arthritis
title_full Development and External Validation of a Model Predicting New‐Onset Chronic Uveitis at Different Disease Durations in Juvenile Idiopathic Arthritis
title_fullStr Development and External Validation of a Model Predicting New‐Onset Chronic Uveitis at Different Disease Durations in Juvenile Idiopathic Arthritis
title_full_unstemmed Development and External Validation of a Model Predicting New‐Onset Chronic Uveitis at Different Disease Durations in Juvenile Idiopathic Arthritis
title_short Development and External Validation of a Model Predicting New‐Onset Chronic Uveitis at Different Disease Durations in Juvenile Idiopathic Arthritis
title_sort development and external validation of a model predicting new‐onset chronic uveitis at different disease durations in juvenile idiopathic arthritis
topic Pediatric Rheumatology
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10108055/
https://www.ncbi.nlm.nih.gov/pubmed/36054539
http://dx.doi.org/10.1002/art.42329
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