Comparing Rituximab and Cyclophosphamide in Induction Therapy for Childhood-Onset Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis: An ARChiVe Registry Cohort Study
Issued Date
2024-01-01
Resource Type
ISSN
2151464X
eISSN
21514658
Scopus ID
2-s2.0-85211624952
Pubmed ID
39467015
Journal Title
Arthritis Care and Research
Rights Holder(s)
SCOPUS
Bibliographic Citation
Arthritis Care and Research (2024)
Suggested Citation
Gagne S.J., Sivaraman V., Bosman E.S., Klamer B., Morishita K.A., Huber A., Orjuela A., Eberhard B., Myrup C., Gerstbacher D., Foell D., Al-Abadi E., McErlane F., Cook K., Wagner-Weiner L., Elder M., Moorthy L.N., Dancey P., Yeung R., Khubchandani R., Deepak S., Charuvanij S., Tarvin S., Shenoi S., Tanner T., Brown K., Cabral D.A. Comparing Rituximab and Cyclophosphamide in Induction Therapy for Childhood-Onset Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis: An ARChiVe Registry Cohort Study. Arthritis Care and Research (2024). doi:10.1002/acr.25455 Retrieved from: https://repository.li.mahidol.ac.th/handle/20.500.14594/102463
Title
Comparing Rituximab and Cyclophosphamide in Induction Therapy for Childhood-Onset Anti-Neutrophil Cytoplasmic Antibody-Associated Vasculitis: An ARChiVe Registry Cohort Study
Author(s)
Gagne S.J.
Sivaraman V.
Bosman E.S.
Klamer B.
Morishita K.A.
Huber A.
Orjuela A.
Eberhard B.
Myrup C.
Gerstbacher D.
Foell D.
Al-Abadi E.
McErlane F.
Cook K.
Wagner-Weiner L.
Elder M.
Moorthy L.N.
Dancey P.
Yeung R.
Khubchandani R.
Deepak S.
Charuvanij S.
Tarvin S.
Shenoi S.
Tanner T.
Brown K.
Cabral D.A.
Sivaraman V.
Bosman E.S.
Klamer B.
Morishita K.A.
Huber A.
Orjuela A.
Eberhard B.
Myrup C.
Gerstbacher D.
Foell D.
Al-Abadi E.
McErlane F.
Cook K.
Wagner-Weiner L.
Elder M.
Moorthy L.N.
Dancey P.
Yeung R.
Khubchandani R.
Deepak S.
Charuvanij S.
Tarvin S.
Shenoi S.
Tanner T.
Brown K.
Cabral D.A.
Author's Affiliation
Great North Children's Hospital
Northwell Health System
SRCC Children’s Hospital
Siriraj Hospital
Seattle Children's Hospital
Nationwide Children’s Hospital
Lucile Packard Children's Hospital Stanford
The Hospital for Sick Children
Akron Children's Hospital
IWK Health Centre
Nottingham University Hospitals NHS Trust
Montefiore Medical Center
Rutgers Robert Wood Johnson Medical School at New Brunswick
Birmingham Children's Hospital
Copenhagen University Hospital
University of Florida
The University of Chicago Medicine
Texas Children's Hospital
BC Children's Hospital
Riley Hospital for Children
UPMC Children’s Hospital of Pittsburgh
The Ohio State University
New Janeway Children s Health and Rehabilitation Centre
University of Münster
Northwell Health System
SRCC Children’s Hospital
Siriraj Hospital
Seattle Children's Hospital
Nationwide Children’s Hospital
Lucile Packard Children's Hospital Stanford
The Hospital for Sick Children
Akron Children's Hospital
IWK Health Centre
Nottingham University Hospitals NHS Trust
Montefiore Medical Center
Rutgers Robert Wood Johnson Medical School at New Brunswick
Birmingham Children's Hospital
Copenhagen University Hospital
University of Florida
The University of Chicago Medicine
Texas Children's Hospital
BC Children's Hospital
Riley Hospital for Children
UPMC Children’s Hospital of Pittsburgh
The Ohio State University
New Janeway Children s Health and Rehabilitation Centre
University of Münster
Corresponding Author(s)
Other Contributor(s)
Abstract
Objective: Granulomatosis with polyangiitis (GPA) and microscopic polyangiitis (MPA) are chronic life-threatening vasculitides requiring substantial immunotherapy. Adult trials identified rituximab (RTX) as an alternative to cyclophosphamide (CYC) for remission induction of GPA and MPA. Disease rarity has limited feasibility of similar trials with pediatric patients. We aim to evaluate the relative efficacy and toxicity of CYC and RTX for patients with childhood GPA and MPA through registry-based comparative evaluation. Methods: From A Registry of Childhood Vasculitis, we identified patients with GPA and MPA who received induction with RTX or CYC. Pediatric Vasculitis Activity Score (PVAS) and Pediatric Vasculitis Damage Index (pVDI) score evaluated disease activity and damage. Descriptive statistics summarized patient characteristics. RTX and CYC comparisons used logistic regression for primary outcomes of postinduction remission (PVAS = 0) or low disease activity (PVAS ≤ 2). Hospital admission for adverse events and pVDI scores were compared using logistic regression and ordinal regression, respectively. Results: Among 104 patients, 43% received RTX, 46% CYC, 11% both. Treatment groups did not significantly differ for diagnosis PVAS and onset age. There was no difference in remission among the groups (63% overall; odds ratio [OR] 1.07, 95% confidence interval [CI] 0.45–2.52). Hospitalizations occurred in 22% of patients receiving RTX versus 10% patients receiving CYC (OR 2.27, 95% CI 0.73–7.05). The median 12-month pVDI score was 1 in both groups (OR 0.98, 95% CI 0.43–2.22). Conclusion: This is the first study comparing CYC and RTX for induction in pediatric GPA and MPA. No significant differences were shown in rates of remission, severe adverse events, or organ damage. Limitations included lack of standardized treatment regimens, retrospectivity, and lack of longitudinal adverse drug-related event data.