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Rituximab as a first-line preventive treatment in pediatric NMOSDs: Preliminary results in 5 children
Giulia Longoni1, Brenda Banwell1, Massimo Filippi1
1Neuroimaging Research Unit, Institute of Experimental Neurology, and Department of Neurology, Division of Neuroscience (G.L., M.F.), San Raffaele Scientific Institute, Vita-Salute San Raffaele University, Milan, Italy; Department of Pediatrics (G.L., E.A.Y.), University of Toronto, and Division of Neurology (G.L., E.A.Y.), The Hospital for Sick Children, Toronto, Ontario, Canada; and Division of Neurology (B.B.), Children's Hospital of Philadelphia, University of Pennsylvania, Philadelphia, PA.
Insights
Rituximab effectively treats childhood neuromyelitis optica spectrum disorders (NMOSDs) by targeting B-cells. This B-cell therapy led to remission, reduced relapse severity, and stabilized disability in pediatric NMOSD patients.
Area of Science:
- Neuroimmunology
- Pediatric Neurology
- Autoimmune Diseases
Background:
- Childhood-onset neuromyelitis optica spectrum disorders (NMOSDs) lack established treatment protocols.
- Neuromyelitis optica immunoglobulin (Ig)G-positive NMOSDs are severe autoimmune conditions affecting the central nervous system.
Purpose of the Study:
- To evaluate the efficacy and safety of a standardized B-cell-targeted immunosuppressive protocol using rituximab as a first-line treatment for pediatric NMOSD patients.
- To assess the impact of rituximab on relapse prevention, disease progression, and neurological disability in children with NMOSDs.
Main Methods:
- Retrospective observational cohort study involving 5 pediatric patients with NMO-IgG-positive NMOSDs.
- Standardized B-cell-targeted immunosuppressive protocol with rituximab for induction and relapse prevention.
Main Results:
- All patients achieved disease remission following rituximab induction.
- Relapses, when they occurred, were associated with CD19(+) B-cell repopulation and were less severe than pre-treatment.
- Neurological disability and MRI findings stabilized or improved in all patients, with manageable side effects.
- Oral steroid discontinuation was achieved in all participants.
Conclusions:
- The rituximab protocol is well-tolerated and demonstrates encouraging results in controlling relapses and disability progression in pediatric NMOSDs.
- Early intervention with rituximab may significantly alter the disease course for children with NMO-IgG-positive NMOSDs.
Objective:
No established therapeutic protocol has been proposed to date for childhood-onset neuromyelitis optica (NMO) spectrum disorders (NMOSDs). We report the response of 5 NMO immunoglobulin (Ig)G-positive pediatric cases to a standardized B-cell-targeted first-line immunosuppressive protocol with rituximab for prevention of relapses.
Methods:
Retrospective observational cohort study.
Results:
All patients included in the study showed disease remission after rituximab induction. Relapses always occurred in conjunction with CD19(+) B-cell repopulation and appeared less severe than prior to treatment. At the end of follow-up, neurologic disability and MRI findings stabilized or improved in all the patients, with only minor and transient side effects. Oral steroid discontinuation was possible in all the patients.
Conclusions:
Our protocol is well-tolerated and has provided encouraging results in terms of control of relapses and progression of disability. An early intervention with rituximab might affect the disease course in pediatric NMO-IgG-positive NMOSDs.
Classification Of Evidence:
This study provides Class IV evidence that for children with NMOSDs, rituximab is well-tolerated and stabilizes or improves neurologic disability.
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