Related Experiment Video
Updated: Jul 6, 2025

A Murine Ommaya Xenograft Model to Study Direct-Targeted Therapy of Leptomeningeal Disease
Published on: January 29, 2021
Control of disease activity with large extended-interval dosing of rituximab/ocrelizumab in highly active pediatric
Melany Venet1,2, Anne Lepine3, Adil Maarouf1
1Department of Neurology, Aix Marseille Univ, APHM, Hôpital de la Timone, CNRS, CRMBM, Marseille, France.
Insights
Extended-interval dosing of rituximab/ocrelizumab (RTX/OCR) is safe and effective for very active pediatric-onset multiple sclerosis (PoMS). A median 18-month interval maintained treatment effectiveness in this pediatric cohort.
Area of Science:
- Neurology
- Immunology
- Pediatrics
Background:
- Adult studies suggest extended-interval dosing of rituximab/ocrelizumab (RTX/OCR) beyond 12 months is safe and potentially improves safety.
- Pediatric-onset multiple sclerosis (PoMS) is a rare and aggressive form of the disease.
Purpose of the Study:
- To evaluate the safety and effectiveness of extended-interval dosing of RTX/OCR in very active PoMS.
- To compare standard 6-month interval dosing with early extended-interval dosing in this population.
Main Methods:
- Observational cohort study of pediatric patients with very active PoMS.
- Patients received either standard 6-month interval RTX/OCR (n=9) or early extended-interval RTX/OCR (n=12, median 18 months).
- Follow-up median of 31 months to assess relapse, disability worsening, and new MRI lesions.
Main Results:
- One patient on standard-interval dosing experienced a relapse.
- No patients showed disability worsening or new T2-weighted lesions.
- Effectiveness of RTX/OCR was maintained with a median extended interval of 18 months.
Conclusions:
- Extended-interval dosing (median 18 months) of RTX/OCR appears safe and effective for very active PoMS.
- This dosing strategy may maintain treatment efficacy while potentially improving safety in pediatric patients.
- Further research is warranted to confirm these findings in larger cohorts.
Abstract:
Recent studies in adults suggested that extended-interval dosing of rituximab/ocrelizumab (RTX/OCR) larger than 12 months was safe and could improve safety. This was an observational cohort study of very active pediatric-onset multiple sclerosis (PoMS) (median (range) age, 16 (12-17) years) treated with RTX/OCR with 6 month standard-interval dosing (n = 9) or early extended-interval dosing (n = 12, median (range) interval 18 months (12-25)). Within a median (range) follow-up of 31 (12-63) months after RTX/OCR onset, one patient (standard-interval) experienced relapse and no patient showed disability worsening or new T2-weighted lesions. This study suggests that the effectiveness of RTX/OCR is maintained with a median extended-interval dosing of 18 months in patients with very active PoMS.

