Jaime Guzman1,2,3, Andrew Henrey4,5,6, Thomas Loughin4,5,6, Roberta A Berard4,5,6, Natalie J Shiff4,5,6, Roman Jurencak4,5,6, Adam M Huber4,5,6, Kiem Oen4,5,6, Kerstin Gerhold4,5,6, Brian M Feldman4,5,6, Rosie Scuccimarri4,5,6, Kristin Houghton4,5,6, Gaëlle Chédeville4,5,6, Kimberly Morishita4,5,6, Bianca Lang4,5,6, Paul Dancey4,5,6, Alan M Rosenberg4,5,6, Julie Barsalou4,5,6, Alessandra Bruns4,5,6, Karen Watanabe Duffy4,5,6, Susanne Benseler4,5,6, Ciaran M Duffy4,5,6, Lori B Tucker4,5,6. 1. From the British Columbia Children's Hospital and the University of British Columbia, Vancouver; Simon Fraser University, Burnaby, British Columbia; London Health Sciences Centre and Western University, London; Children's Hospital of Eastern Ontario and University of Ottawa, Ottawa, Ontario; IWK Health Centre and Dalhousie University, Halifax, Nova Scotia; Winnipeg Children's Hospital and University of Manitoba, Winnipeg, Manitoba; Hospital for Sick Children and University of Toronto, Toronto, Ontario; McGill University Health Centre and McGill University, Montreal, Quebec; Janeway Children's Health and Rehabilitation Centre and Memorial University, Saint John's, Newfoundland and Labrador; Royal University Hospital and University of Saskatchewan, Saskatoon, Saskatchewan; Centre Hospitalier Universitaire Sainte-Justine and Université de Montréal, Montreal; Centre Hospitalier Universitaire de Sherbrooke and Université de Sherbrooke, Sherbrooke, Quebec; Alberta Children's Hospital and University of Calgary, Alberta, Canada; Shands Children's Hospital and University of Florida, Gainesville, Florida, USA. jguzman@cw.bc.ca. 2. The Research in Arthritis in Canadian Children Emphasizing Outcomes cohort was funded by a New Emerging Team research grant from the Canadian Institutes of Health Research (funding reference QNT 69301). Dr. Guzman was funded by a Clinical Investigator Award from the BC Children's Hospital Research Institute, Vancouver, British Columbia, Canada. jguzman@cw.bc.ca. 3. J. Guzman, MD, MSc, Department of Pediatrics, University of British Columbia; A. Henrey, PhD, Department of Statistics and Actuarial Sciences, Simon Fraser University; T. Loughin, PhD, Department of Statistics and Actuarial Sciences, Simon Fraser University; R.A. Berard, MD, MSc, Department of Pediatrics, Western University; N.J. Shiff, MD, MHSc, Department of Pediatrics, University of Florida; R. Jurencak, MD, Department of Pediatrics, University of Ottawa; A.M. Huber, MD, MSc, Department of Pediatrics, Dalhousie University; K. Oen, MD, Department of Pediatrics and Child Health, University of Manitoba; K. Gerhold, MD, Department of Pediatrics and Child Health, University of Manitoba; B.M. Feldman, MD, MSc, Department of Pediatrics and Dalla Lana School of Public Health, University of Toronto; R. Scuccimarri, MD, Research Institute of the McGill University Health Centre; K. Houghton, MD, MSc, Department of Pediatrics, University of British Columbia; G. Chédeville, MD, Research Institute of the McGill University Health Centre; K. Morishita, MD, MHSc, Department of Pediatrics, University of British Columbia; B. Lang, MD, Department of Pediatrics, Dalhousie University; P. Dancey, MD, Department of Pediatrics, Memorial University; A.M. Rosenberg, MD, Department of Pediatrics, University of Saskatchewan; J. Barsalou, MD, MSc, Centre Hospitalier Universitaire Sainte-Justine and Université de Montréal; A. Bruns, MD, MSc, Centre Hospitalier Universitaire de Sherbrooke and Université de Sherbrooke; K. Watanabe Duffy, MD, Department of Pediatrics, University of Ottawa; S. Benseler, MD, PhD, Department of Pediatrics, University of Calgary; C.M. Duffy, MB, MSc, Department of Pediatrics, University of Ottawa; L.B. Tucker, MD, Department of Pediatrics, University of British Columbia. jguzman@cw.bc.ca. 4. From the British Columbia Children's Hospital and the University of British Columbia, Vancouver; Simon Fraser University, Burnaby, British Columbia; London Health Sciences Centre and Western University, London; Children's Hospital of Eastern Ontario and University of Ottawa, Ottawa, Ontario; IWK Health Centre and Dalhousie University, Halifax, Nova Scotia; Winnipeg Children's Hospital and University of Manitoba, Winnipeg, Manitoba; Hospital for Sick Children and University of Toronto, Toronto, Ontario; McGill University Health Centre and McGill University, Montreal, Quebec; Janeway Children's Health and Rehabilitation Centre and Memorial University, Saint John's, Newfoundland and Labrador; Royal University Hospital and University of Saskatchewan, Saskatoon, Saskatchewan; Centre Hospitalier Universitaire Sainte-Justine and Université de Montréal, Montreal; Centre Hospitalier Universitaire de Sherbrooke and Université de Sherbrooke, Sherbrooke, Quebec; Alberta Children's Hospital and University of Calgary, Alberta, Canada; Shands Children's Hospital and University of Florida, Gainesville, Florida, USA. 5. The Research in Arthritis in Canadian Children Emphasizing Outcomes cohort was funded by a New Emerging Team research grant from the Canadian Institutes of Health Research (funding reference QNT 69301). Dr. Guzman was funded by a Clinical Investigator Award from the BC Children's Hospital Research Institute, Vancouver, British Columbia, Canada. 6. J. Guzman, MD, MSc, Department of Pediatrics, University of British Columbia; A. Henrey, PhD, Department of Statistics and Actuarial Sciences, Simon Fraser University; T. Loughin, PhD, Department of Statistics and Actuarial Sciences, Simon Fraser University; R.A. Berard, MD, MSc, Department of Pediatrics, Western University; N.J. Shiff, MD, MHSc, Department of Pediatrics, University of Florida; R. Jurencak, MD, Department of Pediatrics, University of Ottawa; A.M. Huber, MD, MSc, Department of Pediatrics, Dalhousie University; K. Oen, MD, Department of Pediatrics and Child Health, University of Manitoba; K. Gerhold, MD, Department of Pediatrics and Child Health, University of Manitoba; B.M. Feldman, MD, MSc, Department of Pediatrics and Dalla Lana School of Public Health, University of Toronto; R. Scuccimarri, MD, Research Institute of the McGill University Health Centre; K. Houghton, MD, MSc, Department of Pediatrics, University of British Columbia; G. Chédeville, MD, Research Institute of the McGill University Health Centre; K. Morishita, MD, MHSc, Department of Pediatrics, University of British Columbia; B. Lang, MD, Department of Pediatrics, Dalhousie University; P. Dancey, MD, Department of Pediatrics, Memorial University; A.M. Rosenberg, MD, Department of Pediatrics, University of Saskatchewan; J. Barsalou, MD, MSc, Centre Hospitalier Universitaire Sainte-Justine and Université de Montréal; A. Bruns, MD, MSc, Centre Hospitalier Universitaire de Sherbrooke and Université de Sherbrooke; K. Watanabe Duffy, MD, Department of Pediatrics, University of Ottawa; S. Benseler, MD, PhD, Department of Pediatrics, University of Calgary; C.M. Duffy, MB, MSc, Department of Pediatrics, University of Ottawa; L.B. Tucker, MD, Department of Pediatrics, University of British Columbia.
Abstract
OBJECTIVE: To estimate the probability of early remission with conventional treatment for each child with juvenile idiopathic arthritis (JIA). Children with a low chance of remission may be candidates for initial treatment with biologics or triple disease-modifying antirheumatic drugs (DMARD). METHODS: We used data from 1074 subjects in the Research in Arthritis in Canadian Children emphasizing Outcomes (ReACCh-Out) cohort. The predicted outcome was clinically inactive disease for ≥ 6 months starting within 1 year of JIA diagnosis in patients who did not receive early biologic agents or triple DMARD. Models were developed in 200 random splits of 75% of the cohort and tested on the remaining 25% of subjects, calculating expected and observed frequencies of remission and c-index values. RESULTS: Our best Cox logistic model combining 18 clinical variables a median of 2 days after diagnosis had a c-index of 0.69 (95% CI 0.67-0.71), better than using JIA category alone (0.59, 95% CI 0.56-0.63). Children in the lowest probability decile had a 20% chance of remission and 21% attained remission; children in the highest decile had a 69% chance of remission and 73% attained remission. Compared to 5% of subjects identified by JIA category alone, the model identified 14% of subjects as low chance of remission (probability < 0.25), of whom 77% failed to attain remission. CONCLUSION: Although the model did not meet our a priori performance threshold (c-index > 0.70), it identified 3 times more subjects with low chance of remission than did JIA category alone, and it may serve as a benchmark for assessing value added by future laboratory/imaging biomarkers.
OBJECTIVE: To estimate the probability of early remission with conventional treatment for each child with juvenile idiopathic arthritis (JIA). Children with a low chance of remission may be candidates for initial treatment with biologics or triple disease-modifying antirheumatic drugs (DMARD). METHODS: We used data from 1074 subjects in the Research in Arthritis in Canadian Children emphasizing Outcomes (ReACCh-Out) cohort. The predicted outcome was clinically inactive disease for ≥ 6 months starting within 1 year of JIA diagnosis in patients who did not receive early biologic agents or triple DMARD. Models were developed in 200 random splits of 75% of the cohort and tested on the remaining 25% of subjects, calculating expected and observed frequencies of remission and c-index values. RESULTS: Our best Cox logistic model combining 18 clinical variables a median of 2 days after diagnosis had a c-index of 0.69 (95% CI 0.67-0.71), better than using JIA category alone (0.59, 95% CI 0.56-0.63). Children in the lowest probability decile had a 20% chance of remission and 21% attained remission; children in the highest decile had a 69% chance of remission and 73% attained remission. Compared to 5% of subjects identified by JIA category alone, the model identified 14% of subjects as low chance of remission (probability < 0.25), of whom 77% failed to attain remission. CONCLUSION: Although the model did not meet our a priori performance threshold (c-index > 0.70), it identified 3 times more subjects with low chance of remission than did JIA category alone, and it may serve as a benchmark for assessing value added by future laboratory/imaging biomarkers.
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
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: Pamela F Weiss; Timothy G Brandon; Meghan E Ryan; Erin B Treemarcki; Stephanie Armendariz; Tracey B Wright; Chetna Godiwala; Matthew L Stoll; Rui Xiao; Daniel Lovell Journal: Arthritis Care Res (Hoboken) Date: 2021-08-07 Impact factor: 5.178
Authors: Patricia Vega-Fernandez; Tracy V Ting; Edward J Oberle; Courtney McCracken; Janet Figueroa; Mekibib Altaye; Amy Cassedy; Gurjit S Kaeley; Johannes Roth Journal: Arthritis Care Res (Hoboken) Date: 2021-07-30 Impact factor: 5.178
Authors: Andrew Henrey; Veronika Rypdal; Martin Rypdal; Thomas Loughin; Ellen Nordal; Jaime Guzman Journal: Arthritis Res Ther Date: 2020-01-15 Impact factor: 5.156