Abstract
Original language | English |
---|---|
Pages (from-to) | 567-574 |
Number of pages | 8 |
Journal | Journal of rheumatology |
Volume | 48 |
Issue number | 4 |
DOIs | |
Publication status | Published - 1 Apr 2021 |
Keywords
- Autoinflammation
- Cytokine inhibitors
- Inflammatory bowel disease
- Pediatric rheumatology
- Systemic juvenile idiopathic arthritis
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In: Journal of rheumatology, Vol. 48, No. 4, 01.04.2021, p. 567-574.
Research output: Contribution to journal › Article › Academic › peer-review
TY - JOUR
T1 - Inflammatory bowel disease in children with systemic juvenile idiopathic arthritis
AU - Maller, Justine
AU - Fox, Emily
AU - Park, K. T.
AU - Paul, Sarah Sertial
AU - Baszis, Kevin
AU - Borocco, Charlotte
AU - Prahalad, Sampath
AU - Quartier, Pierre
AU - Reinhardt, Adam
AU - Schonenberg-Meinema, Dieneke
AU - Shipman-Duensing, Lauren
AU - Terreri, Maria Teresa
AU - Simard, Julia
AU - Lavi, Idit
AU - Chalom, Elizabeth
AU - Hsu, Joyce
AU - Zisman, Devy
AU - Mellins, Elizabeth D.
AU - CARRA Legacy Registry Investigators
AU - Abramson, L.
AU - Anderson, E.
AU - Andrew, M.
AU - Battle, N.
AU - Becker, M.
AU - Benham, H.
AU - Beukelman, T.
AU - Birmingham, J.
AU - Blier, P.
AU - Brown, A.
AU - Brunner, H.
AU - Cabrera, A.
AU - Canter, D.
AU - Carlton, D.
AU - Caruso, B.
AU - Ceracchio, L.
AU - Chang, J.
AU - Charpentier, P.
AU - Clark, K.
AU - Dean, J.
AU - Dedeoglu, F.
AU - Feldman, B.
AU - Ferguson, P.
AU - Fox, M.
AU - Francis, K.
AU - Gervasini, M.
AU - Goldsmith, D.
AU - Gorton, G.
AU - Gottlieb, B.
AU - Graham, T.
AU - Griffin, T.
AU - Grosbein, H.
N1 - Funding Information: This study was supported by the National Institutes of Health (NIH; grant T32AR050942-14; JM); Tashia and John Morgridge Endowed Postdoctoral Fellow Clinical Trainee Award, Stanford Maternal & Child Health Research Institute (JM); The Marcus Foundation Inc., Atlanta, Georgia (SP); Feldman Family Foundation Visiting Professors Program, Stanford University School of Medicine (DZ); Arthritis Foundation Great Western Region Center of Excellence for Arthritis (EDM); The Lucile Packard Foundation for Children?s Health (EDM). The Childhood Arthritis and Rheumatology Research Alliance (CARRA) Legacy Registry was supported by a grant from National Institute of Arthritis and Musculoskeletal and Skin Diseases of the NIH under award Number RC2AR058934. The content is solely the responsibility of the authors and does not necessarily represent the official views of the NIH. The CARRA Legacy Registry was also supported by CARRA, Friends of CARRA, the Arthritis Foundation, and the Duke Clinical Research Institute. Funding Information: This study was supported by the National Institutes of Health (NIH; grant T32AR050942-14; JM); Tashia and John Morgridge Endowed Postdoctoral Fellow Clinical Trainee Award, Stanford Maternal & Child Health Research Institute (JM); The Marcus Foundation Inc., Atlanta, Georgia (SP); Feldman Family Foundation Visiting Professors Program, Stanford University School of Medicine (DZ); Arthritis Foundation Great Western Region Center of Excellence for Arthritis (EDM); The Lucile Packard Foundation for Children’s Health (EDM). The Childhood Arthritis and Rheumatology Research Alliance (CARRA) Legacy Registry was supported by a grant from National Institute of Arthritis and Musculoskeletal and Skin Diseases of the NIH under award Number RC2AR058934. The content is solely the responsibility of the authors and does not necessarily represent the official views of the NIH. The CARRA Legacy Registry was also supported by CARRA, Friends of CARRA, the Arthritis Foundation, and the Duke Clinical Research Institute. 1J. Maller, MD, PhD, Department of Pediatrics, Division of Rheumatology, Stanford University School of Medicine, Stanford, California, USA; 2E. Fox, MD, Department of Pediatrics, Division of Rheumatology, Stanford University School of Medicine, Stanford, California, and Department of Pediatrics, Division of Rheumatology, Children’s Mercy Hospital, University of Missouri–Kansas City, Kansas City, Missouri, USA; 3K.T. Park, MD, Department of Pediatrics, Division of Gastroenterology, Stanford University School of Medicine, Stanford, California, USA; 4S. Sertial Paul, DO, Department of Pediatrics, Goryeb Children’s Hospital, Morristown, New Jersey, USA; 5K. Baszis, MD, Department of Pediatrics, Washington University School of Medicine, St. Louis, Missouri, USA; 6C. Borocco, MD, Paris University, Imagine Institute and Pediatric Hematology-Immunology and Rheumatology Unit, Necker Hospital, AP-HP, Paris, France; 7S. Prahalad, MD, Department of Pediatrics and Department of Genetics, Emory University School of Medicine, Children’s Healthcare of Atlanta, Atlanta, Georgia, USA; 8P. Quartier, MD, Paris University, Imagine Institute, RAISE Reference Centre and Pediatric Hematology-Immunology and Rheumatology Unit, Necker Hospital, AP-HP, Paris, France; 9A. Reinhardt, MD, Department of Pediatrics, Boys Town National Research Hospital, Omaha, Nebraska, USA; 10D. Schonenberg-Meinema, MD, Department of Pediatric Immunology, Rheumatology and Infectious Diseases, Amsterdam Universitair Medische Centra, Amsterdam, the Netherlands; 11L. Shipman-Duensing, MD, Department of Pediatrics, Division of Rheumatology, University of Alabama at Birmingham, Birmingham, Alabama, USA; 12M.T. Terreri, MD, Department of Pediatrics, Pediatric Rheumatology Unit, Universidade Federal de São Paulo, São Paulo, Brazil; 13J. Simard, ScD, Department of Health Research & Policy, Division of Epidemiology, and Department of Medicine, Division of Immunology & Rheumatology, Stanford University, Stanford, California, USA; 14I. Lavi, MA, Department of Community Medicine and Epidemiology, Carmel Medical Center, Haifa, Israel; 15E. Chalom, MD, Department of Pediatrics, Publisher Copyright: © 2021 Journal of Rheumatology. All rights reserved. Copyright: Copyright 2021 Elsevier B.V., All rights reserved.
PY - 2021/4/1
Y1 - 2021/4/1
N2 - Objective. The incidence of inflammatory bowel disease (IBD) in juvenile idiopathic arthritis (JIA) is higher than in the general pediatric population. However, reports of IBD in the systemic JIA (sJIA) subtype are limited. We sought to characterize sJIA patients diagnosed with IBD and to identify potential contributing risk factors. Methods. Using an internationally distributed survey, we identified 16 patients with sJIA who were subsequently diagnosed with IBD (sJIA-IBD cohort). Five hundred twenty-two sJIA patients without IBD were identified from the CARRA Legacy Registry and served as the sJIA-only cohort for comparison. Differences in demographic, clinical characteristics, and therapy were assessed using chi-square test, Fisher exact test, t-test, and univariate and multivariate logistic regression, as appropriate. Results. Of the patients with sJIA-IBD, 75% had a persistent sJIA course and 25% had a history of macrophage activation syndrome. sJIA-IBD subjects were older at sJIA diagnosis, more often non-White, had a higher rate of IBD family history, and were more frequently treated with etanercept or canakinumab compared to sJIA-only subjects. Sixty-nine percent of sJIA-IBD patients successfully discontinued sJIA medications following IBD diagnosis, and sJIA symptoms resolved in 9 of 12 patients treated with tumor necrosis factor-α (TNF-α) inhibitors. Conclusion. IBD in the setting of sJIA is a rare occurrence. The favorable response of sJIA symptoms to therapeutic TNF-α inhibition suggests that the sJIA-IBD cohort may represent a mechanistically distinct sJIA subgroup. Our study highlights the importance of maintaining a high level of suspicion for IBD when gastrointestinal involvement occurs in patients with sJIA and the likely broad benefit of TNF-α inhibition in those cases.
AB - Objective. The incidence of inflammatory bowel disease (IBD) in juvenile idiopathic arthritis (JIA) is higher than in the general pediatric population. However, reports of IBD in the systemic JIA (sJIA) subtype are limited. We sought to characterize sJIA patients diagnosed with IBD and to identify potential contributing risk factors. Methods. Using an internationally distributed survey, we identified 16 patients with sJIA who were subsequently diagnosed with IBD (sJIA-IBD cohort). Five hundred twenty-two sJIA patients without IBD were identified from the CARRA Legacy Registry and served as the sJIA-only cohort for comparison. Differences in demographic, clinical characteristics, and therapy were assessed using chi-square test, Fisher exact test, t-test, and univariate and multivariate logistic regression, as appropriate. Results. Of the patients with sJIA-IBD, 75% had a persistent sJIA course and 25% had a history of macrophage activation syndrome. sJIA-IBD subjects were older at sJIA diagnosis, more often non-White, had a higher rate of IBD family history, and were more frequently treated with etanercept or canakinumab compared to sJIA-only subjects. Sixty-nine percent of sJIA-IBD patients successfully discontinued sJIA medications following IBD diagnosis, and sJIA symptoms resolved in 9 of 12 patients treated with tumor necrosis factor-α (TNF-α) inhibitors. Conclusion. IBD in the setting of sJIA is a rare occurrence. The favorable response of sJIA symptoms to therapeutic TNF-α inhibition suggests that the sJIA-IBD cohort may represent a mechanistically distinct sJIA subgroup. Our study highlights the importance of maintaining a high level of suspicion for IBD when gastrointestinal involvement occurs in patients with sJIA and the likely broad benefit of TNF-α inhibition in those cases.
KW - Autoinflammation
KW - Cytokine inhibitors
KW - Inflammatory bowel disease
KW - Pediatric rheumatology
KW - Systemic juvenile idiopathic arthritis
UR - http://www.scopus.com/inward/record.url?scp=85104818803&partnerID=8YFLogxK
U2 - https://doi.org/10.3899/JRHEUM.200230
DO - https://doi.org/10.3899/JRHEUM.200230
M3 - Article
C2 - 32541073
SN - 0315-162X
VL - 48
SP - 567
EP - 574
JO - Journal of rheumatology
JF - Journal of rheumatology
IS - 4
ER -