TY - JOUR
T1 - Establishing an updated core domain set for studies in juvenile idiopathic arthritis
T2 - A report from the OMERACT 2018 JIA workshop
AU - Morgan, Esi M.
AU - Munro, Jane E.
AU - Horonjeff, Jennifer
AU - Horgan, Ben
AU - Shea, Beverley
AU - Feldman, Brian M.
AU - Clairman, Hayyah
AU - Bingham, Clifton O.
AU - Thornhill, Susan
AU - Strand, Vibeke
AU - Alongi, Alessandra
AU - Magni-Manzoni, Silvia
AU - Van Rossum, Marion A.J.
AU - Vesely, Richard
AU - Vojinovic, Jelena
AU - Brunner, Hermine I.
AU - Harris, Julia G.
AU - Horton, Daniel B.
AU - Lovell, Daniel J.
AU - Mannion, Melissa
AU - Rahimi, Homaira
AU - Ravelli, Angelo
AU - Ringold, Sarah
AU - Ruperto, Nicolino
AU - Schrandt, M. Suzanne
AU - Shenoi, Susan
AU - Shiff, Natalie J.
AU - Toupin-April, Karine
AU - Tzaribachev, Nikolay
AU - Weiss, Pamela
AU - Consolaro, Alessandro
N1 - Funding Information:
OMERACT (Outcome Measures in Rheumatology) is an international health outcomes measurement group, which has been funded by unrestricted support from more than 12 pharmaceutical and clinical research organizations in the last 2 years. Drs. Bingham and Strand are members of the executive committee for OMERACT, but receive no financial remuneration for their efforts. OMERACT funded 2 patient research partners (BH, JH) to participate at OMERACT 2018. The online discussion boards (ODB) in Australia were supported with funding of Arthritis Australia. The ODB in Italy were supported by AlfaSigma SpA. The ODB in the United States were funded in part by the Arthritis Foundation and the Patient-Centered Outcomes Research Institute (PPRN-1306-04601). Conduct of the Delphi survey was funded by the Ho Family Research Chair in Autoimmune Diseases, awarded to Dr. Feldman. E.M. Morgan, MD, Associate Professor, Division of Rheumatology, Cincinnati Children’s Hospital Medical Center, University of Cincinnati; J.E. Munro, MBBS, Associate Professor, Head of Rheumatology at the Royal Children’s Hospital, and Group Leader, Arthritis research, Murdoch Children’s Research Institute; J. Horonjeff, PhD, Instructor, Office of Research, Division of Rheumatology, Columbia University Medical Center; B. Horgan, Consumer Advocate, Consumer and Community Health Research Network; B. Shea, PhD, Clinical Investigator, Ottawa Hospital Research Institute, and Adjunct Professor, School of Epidemiology and Public Health, University of Ottawa; B.M. Feldman, MD, MSc, FRCPC, Professor, Pediatrics, Medicine, Institute of Health Policy Management and Evaluation, University of Toronto, and Head, Division of Rheumatology, The Hospital for Sick Children; H. Clairman, MSc, Clinical Research Project Coordinator, Child Health Evaluative Sciences, Hospital for Sick Children; C.O. Bingham III, MD, Professor of Medicine, Division of Rheumatology, Johns Hopkins University; S. Thornhill, AA, Qualitative Research Consultant, Thornhill Associates; V. Strand, MD, Biopharmaceutical Consultant; A. Alongi, PhD Student, IRCCS Istituto Giannina Gaslini, Clinica Pediatrica e Reumatologia, and Università degli studi di Genova; S. Magni-Manzoni, MD, Rheumatology Division, Ospedale Pediatrico Bambino Gesù; M.A. van Rossum, MD, PhD, Pediatric Rheumatologist/Immunologist, Amsterdam Rheumatology and Immunology Center/Reade l Emma Children’s Hospital, Amsterdam Medical Center; R. Vesely, MD, Head of the Rheumatology, Respiratory, Gastroenterology and Immunology Office Scientific and Regulatory Management Department, European Medicines Agency; J. Vojinovic, MD, PhD, Professor, University of Nis, Faculty of Medicine, Department of Pediatric Rheumatology; H.I. Brunner, Professor, Division of Rheumatology, Cincinnati Children’s Hospital Medical Center; J.G. Harris, MD, Assistant Professor, Division of Rheumatology, Children’s Mercy; D.B. Horton, MD, MSCE, Assistant Professor, Division of Pediatric Rheumatology, Rutgers Robert Wood Johnson Medical School, Institute for Health, Health Care Policy and Aging Research; D.J. Lovell, MD, Professor, Division of Rheumatology, Cincinnati Children’s Hospital Medical Center; M. Mannion, MD, Assistant Professor of Rheumatology, University of Alabama at Birmingham; H. Rahimi, MD, Assistant Professor, Division of Rheumatology, University of Rochester, Golisano Children’s Hospital; A. Ravelli, MD, Professor of Pediatrics, Università degli Studi di Genova and IRCCS Istituto Giannina Gaslini; S. Ringold, MD, Assistant Professor, Division of Rheumatology, Seattle Children’s Hospital; N. Ruperto, MD, MPH, IRCCS Istituto Giannina Gaslini, Clinica Pediatrica e Reumatologia, PRINTO; M.S. Schrandt, JD, Director, Patient Engagement, Arthritis Foundation; S. Shenoi, MBBS, MS Associate Professor, Seattle Children’s Hospital; N.J. Shiff, MD, University of Florida, Shands Children’s Hospital; K. Toupin-April, PhD, Associate Scientist, Children’s Hospital of Eastern Ontario Research Institute, and Assistant Professor, Department of Pediatrics and School of Rehabilitation Sciences, University of Ottawa; N. Tzaribachev, MD, Pediatric Rheumatology Research Institute; P. Weiss, MD, MSCE, Associate Professor, Division of Rheumatology, Children’s Hospital of Philadelphia; A. Consolaro, MD, PhD, Assistant Professor, IRCCS Istituto Giannina Gaslini, Clinica Pediatrica e Reumatologia, and Università degli studi di Genova. Address correspondence to Dr. E.M. Morgan, Associate Professor of Pediatrics, Division of Rheumatology, Cincinnati Children’s Hospital Medical Center, 3333 Burnet Ave., MLC 4010, Cincinnati, Ohio 45229, USA. E-mail: [email protected] Accepted for publication January 24, 2019.
Publisher Copyright:
The Journal of Rheumatology Copyright © 2019. All rights reserved.
PY - 2019/8/1
Y1 - 2019/8/1
N2 - Objective. The current Juvenile Idiopathic Arthritis (JIA) Core Set used in randomized controlled trials (RCT) and longitudinal observational studies (LOS) was developed without the input of patients/parents. At the Outcome Measures in Rheumatology (OMERACT) 2016, a special interest group voted to reconsider the core set, incorporating broader input. We describe subsequent work culminating in an OMERACT 2018 plenary and consensus voting. Methods. Candidate domains were identified through literature review, qualitative surveys, and online discussion boards (ODB) held with patients with JIA and parents in Australia, Italy, and the United States. A Delphi process with parents, patients, healthcare providers, researchers, and regulators served to edit the domain list and prioritize candidate domains. After the presentation of results, OMERACT workshop participants voted, with consensus set at > 70%. Results. Participants in ODB were 53 patients with JIA (ages 15–24 yrs) and 55 parents. Three rounds of Delphi considering 27 domains were completed by 190 (response rate 85%), 201 (84%), and 182 (77%) people, respectively, from 50 countries. There was discordance noted between domains prioritized by patients/parents compared to others. OMERACT conference voting approved domains for JIA RCT and LOS with 83% endorsement. Mandatory domains are pain, joint inflammatory signs, activity limitation/physical function, patient’s perception of disease activity (overall well-being), and adverse events. Mandatory in specific circumstances: inflammation/other features relevant to specific JIA categories. Conclusion. Following the OMERACT methodology, we developed an updated JIA Core Domain Set. Next steps are to identify and systematically evaluate best outcome measures for these domains.
AB - Objective. The current Juvenile Idiopathic Arthritis (JIA) Core Set used in randomized controlled trials (RCT) and longitudinal observational studies (LOS) was developed without the input of patients/parents. At the Outcome Measures in Rheumatology (OMERACT) 2016, a special interest group voted to reconsider the core set, incorporating broader input. We describe subsequent work culminating in an OMERACT 2018 plenary and consensus voting. Methods. Candidate domains were identified through literature review, qualitative surveys, and online discussion boards (ODB) held with patients with JIA and parents in Australia, Italy, and the United States. A Delphi process with parents, patients, healthcare providers, researchers, and regulators served to edit the domain list and prioritize candidate domains. After the presentation of results, OMERACT workshop participants voted, with consensus set at > 70%. Results. Participants in ODB were 53 patients with JIA (ages 15–24 yrs) and 55 parents. Three rounds of Delphi considering 27 domains were completed by 190 (response rate 85%), 201 (84%), and 182 (77%) people, respectively, from 50 countries. There was discordance noted between domains prioritized by patients/parents compared to others. OMERACT conference voting approved domains for JIA RCT and LOS with 83% endorsement. Mandatory domains are pain, joint inflammatory signs, activity limitation/physical function, patient’s perception of disease activity (overall well-being), and adverse events. Mandatory in specific circumstances: inflammation/other features relevant to specific JIA categories. Conclusion. Following the OMERACT methodology, we developed an updated JIA Core Domain Set. Next steps are to identify and systematically evaluate best outcome measures for these domains.
KW - Clinical trials
KW - Juvenile idiopathic arthritis
KW - OMERACT
KW - Outcome assessment
KW - Quality of life
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U2 - 10.3899/jrheum.181088
DO - 10.3899/jrheum.181088
M3 - Article
C2 - 30770499
AN - SCOPUS:85070189192
SN - 0315-162X
VL - 46
SP - 1006
EP - 1013
JO - Journal of Rheumatology
JF - Journal of Rheumatology
IS - 8
ER -