Jaime Guzman1,2, Andrew Henrey3,4, Thomas Loughin3,4, Roberta A Berard3,4, Natalie J Shiff3,4, Roman Jurencak3,4, Susanne M Benseler3,4, Lori B Tucker. 1. From the British Columbia Children's Hospital and the University of British Columbia, Vancouver; Simon Fraser University, Burnaby, British Columbia; the Alberta Children's Hospital and University of Calgary, Calgary, Alberta; London Health Sciences Centre and Western University, London; the Children's Hospital of Eastern Ontario and University of Ottawa, Ottawa, Ontario, Canada; the Shands Children's Hospital and University of Florida, Gainesville, Florida, USA. jguzman@cw.bc.ca. 2. J. Guzman, MD, MSc, British Columbia Children's Hospital and the University of British Columbia; A. Henrey, PhD, Simon Fraser University; T. Loughin, PhD, Professor, Simon Fraser University; R.A. Berard, MD, MSc, London Health Sciences Centre and Western University; N.J. Shiff, MD, MSc, Shands Children's Hospital and University of Florida; R. Jurencak, MD, Children's Hospital of Eastern Ontario and University of Ottawa; S.M. Benseler, MD, PhD, Alberta Children's Hospital and University of Calgary; L.B. Tucker, MD, British Columbia Children's Hospital and the University of British Columbia. jguzman@cw.bc.ca. 3. From the British Columbia Children's Hospital and the University of British Columbia, Vancouver; Simon Fraser University, Burnaby, British Columbia; the Alberta Children's Hospital and University of Calgary, Calgary, Alberta; London Health Sciences Centre and Western University, London; the Children's Hospital of Eastern Ontario and University of Ottawa, Ottawa, Ontario, Canada; the Shands Children's Hospital and University of Florida, Gainesville, Florida, USA. 4. J. Guzman, MD, MSc, British Columbia Children's Hospital and the University of British Columbia; A. Henrey, PhD, Simon Fraser University; T. Loughin, PhD, Professor, Simon Fraser University; R.A. Berard, MD, MSc, London Health Sciences Centre and Western University; N.J. Shiff, MD, MSc, Shands Children's Hospital and University of Florida; R. Jurencak, MD, Children's Hospital of Eastern Ontario and University of Ottawa; S.M. Benseler, MD, PhD, Alberta Children's Hospital and University of Calgary; L.B. Tucker, MD, British Columbia Children's Hospital and the University of British Columbia.
Abstract
OBJECTIVE: 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. METHODS: 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, r2, 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. RESULTS: 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. CONCLUSION: 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.
OBJECTIVE: 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. METHODS: 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, r2, 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. RESULTS: 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. CONCLUSION: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.
Authors: Elham Rezaei; Daniel Hogan; Brett Trost; Anthony J Kusalik; Gilles Boire; David A Cabral; Sarah Campillo; Gaëlle Chédeville; Anne-Laure Chetaille; Paul Dancey; Ciaran Duffy; Karen Watanabe Duffy; John Gordon; Jaime Guzman; Kristin Houghton; Adam M Huber; Roman Jurencak; Bianca Lang; Kimberly Morishita; Kiem G Oen; Ross E Petty; Suzanne E Ramsey; Rosie Scuccimarri; Lynn Spiegel; Elizabeth Stringer; Regina M Taylor-Gjevre; Shirley M L Tse; Lori B Tucker; Stuart E Turvey; Susan Tupper; Rae S M Yeung; Susanne Benseler; Janet Ellsworth; Chantal Guillet; Chandima Karananayake; Nazeem Muhajarine; Johannes Roth; Rayfel Schneider; Alan M Rosenberg Journal: Rheumatology (Oxford) Date: 2020-09-01 Impact factor: 7.580
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Authors: Stephanie J W Shoop-Worrall; Kimme L Hyrich; Suzanne M M Verstappen; Jamie C Sergeant; Eileen Baildam; Alice Chieng; Joyce Davidson; Helen Foster; Yiannis Ioannou; Flora McErlane; Lucy R Wedderburn; Wendy Thomson; Janet E McDonagh Journal: Arthritis Care Res (Hoboken) Date: 2020-04 Impact factor: 4.794
Authors: Veronika Rypdal; Jaime Guzman; Andrew Henrey; Thomas Loughin; Mia Glerup; Ellen Dalen Arnstad; Kristiina Aalto; Marite Rygg; Susan Nielsen; Troels Herlin; Anders Fasth; Lillemor Berntson; Martin Rypdal; Ellen Nordal Journal: Arthritis Res Ther Date: 2019-12-05 Impact factor: 5.156
Authors: Veronika Rypdal; Ellen Dalen Arnstad; Kristiina Aalto; Lillemor Berntson; Maria Ekelund; Anders Fasth; Mia Glerup; Troels Herlin; Susan Nielsen; Suvi Peltoniemi; Marek Zak; Marite Rygg; Martin Rypdal; Ellen Nordal Journal: Arthritis Res Ther Date: 2018-05-03 Impact factor: 5.156