Comparative Effectiveness of Disease-Modifying Treatments in Double Seronegative Neuromyelitis Optica Spectrum Disorder
Issued Date
2026-03-01
Resource Type
eISSN
23327812
Scopus ID
2-s2.0-105029525272
Pubmed ID
41637688
Journal Title
Neurology R Neuroimmunology Neuroinflammation
Volume
13
Issue
2
Rights Holder(s)
SCOPUS
Bibliographic Citation
Neurology R Neuroimmunology Neuroinflammation Vol.13 No.2 (2026) , e200514
Suggested Citation
Mahler J.V., Vallejos G.B., Mikami T., Bilodeau P.A., Anderson M., Drosu N., Bobrowski-Khoury N., Silva G.D., Solti M., Apóstolos-Pereira S.L., Callegaro D., Leles Vieira de Souza B., Manzano G.S., Vishnevetsky A., Gillani R., Pasquale O., Kim A., Vij R., Kister I., Gibbons E.L., Jacob A., Huda S., Said Y., Krett J.D., Sotirchos E.S., Ramprasad M., Abboud H., Crelier V.T.C., Dos Santos G., Uawithya E., Siritho S., Sezen A., Altintas A., Gai F., Guo Y., Bhattacharyya S., Levy M., Matiello M. Comparative Effectiveness of Disease-Modifying Treatments in Double Seronegative Neuromyelitis Optica Spectrum Disorder. Neurology R Neuroimmunology Neuroinflammation Vol.13 No.2 (2026) , e200514. doi:10.1212/NXI.0000000000200514 Retrieved from: https://repository.li.mahidol.ac.th/handle/123456789/115062
Title
Comparative Effectiveness of Disease-Modifying Treatments in Double Seronegative Neuromyelitis Optica Spectrum Disorder
Author(s)
Mahler J.V.
Vallejos G.B.
Mikami T.
Bilodeau P.A.
Anderson M.
Drosu N.
Bobrowski-Khoury N.
Silva G.D.
Solti M.
Apóstolos-Pereira S.L.
Callegaro D.
Leles Vieira de Souza B.
Manzano G.S.
Vishnevetsky A.
Gillani R.
Pasquale O.
Kim A.
Vij R.
Kister I.
Gibbons E.L.
Jacob A.
Huda S.
Said Y.
Krett J.D.
Sotirchos E.S.
Ramprasad M.
Abboud H.
Crelier V.T.C.
Dos Santos G.
Uawithya E.
Siritho S.
Sezen A.
Altintas A.
Gai F.
Guo Y.
Bhattacharyya S.
Levy M.
Matiello M.
Vallejos G.B.
Mikami T.
Bilodeau P.A.
Anderson M.
Drosu N.
Bobrowski-Khoury N.
Silva G.D.
Solti M.
Apóstolos-Pereira S.L.
Callegaro D.
Leles Vieira de Souza B.
Manzano G.S.
Vishnevetsky A.
Gillani R.
Pasquale O.
Kim A.
Vij R.
Kister I.
Gibbons E.L.
Jacob A.
Huda S.
Said Y.
Krett J.D.
Sotirchos E.S.
Ramprasad M.
Abboud H.
Crelier V.T.C.
Dos Santos G.
Uawithya E.
Siritho S.
Sezen A.
Altintas A.
Gai F.
Guo Y.
Bhattacharyya S.
Levy M.
Matiello M.
Author's Affiliation
Harvard Medical School
Universidade de São Paulo
Johns Hopkins University School of Medicine
University of Liverpool
NYU Grossman School of Medicine
CASE School of Medicine
Universidade Federal Fluminense
University Hospitals Case Medical Center
Koç University
Siriraj Hospital
Beijing Tongren Hospital, Capital Medical University
Creighton University School of Medicine
Cleveland Clinic Abu Dhabi
The Walton Centre NHS Foundation Trust
Universidade de São Paulo
Johns Hopkins University School of Medicine
University of Liverpool
NYU Grossman School of Medicine
CASE School of Medicine
Universidade Federal Fluminense
University Hospitals Case Medical Center
Koç University
Siriraj Hospital
Beijing Tongren Hospital, Capital Medical University
Creighton University School of Medicine
Cleveland Clinic Abu Dhabi
The Walton Centre NHS Foundation Trust
Corresponding Author(s)
Other Contributor(s)
Abstract
BACKGROUND AND OBJECTIVES: Double seronegative NMOSD (DS-NMOSD) lacks approved disease-modifying treatments, and limited data exist on optimal relapse-prevention strategies. In this multicenter, international, retrospective cohort study, we sought to compare the real-world effectiveness of anti-CD20 agents vs nonspecific immunosuppressants as disease-modifying strategies for relapse prevention in DS-NMOSD. METHODS: A retrospective cohort database was constructed using standardized data collection from medical records across collaborating centers in the United States, Brazil, the United Kingdom, Thailand, Turkiye, and China. Patients meeting IPND-2015 NMOSD criteria with negative serum aquaporin-4 and myelin oligodendrocyte glycoprotein antibody testing via cell-based assays and at least 12 months of follow-up were reviewed. The primary outcome was the incidence rate ratio (IRR) of relapses; secondary outcomes included the annualized relapse rate (ARR) and time to relapse. RESULTS: A total of 103 patients with DS-NMOSD met study criteria, with a median follow-up of 6 years. Anti-CD20 therapy was associated with a significantly lower IRR (0.02, 95% CI 0.01-0.04) and ARR (0.17, 95% CI 0.07-0.40) compared with nonspecific immunosuppressants (0.76, 95% CI 0.40-1.43) after adjusting for covariates. Survival analysis demonstrated a prolonged relapse-free interval with anti-CD20 agents. DISCUSSION: Our findings support the use of B-cell depletion as a potentially superior relapse-prevention strategy in DS-NMOSD, highlighting its potential as a first-line therapy. CLASSIFICATION OF EVIDENCE: This study provides Class IV evidence that, in patients with DS-NMOSD, treatment with a DMT reduces relapse incidence rate ratio compared with no treatment and anti-CD20 DMTs are associated with a lower relapse incidence rate ratio compared with nonspecific immunosuppressants.