Myelin-oligodendrocyte glycoprotein antibody-associated disorder (MOGAD) is an emerging disease entity related to a broad spectrum of acquired inflammatory demyelination of the central nervous system. Previous studies have demonstrated the efficacy of rituximab (RTX), an anti-CD20 monoclonal antibody, in neuromyelitis optica spectrum disorder, and multiple sclerosis. Although RTX is commonly used in MOGAD as well, the benefit of this therapy is still underinvestigated.
To evaluate the effect of RTX in adult and pediatric MOGAD including the change in annualized relapse rate (ARR), and expanded disability status (EDSS), the post-treatment ARR, as well as the prevalence of freedom-from-relapse and EDSS progression.
A literature search was conducted on MEDLINE, EMBASE, and Cochrane databases. Eligibility criteria included observational studies that evaluated the efficacy of RTX in adult and pediatric MOGAD. We excluded review articles, case reports, studies that included fewer than 2 patients, and unpublished studies. We performed meta-analysis using Comprehensive Meta-analysis version 3.3 software from Biostat, Inc (Eaglewood, NJ, USA). Study results were statistically combined using a random-effect model and displayed with forest plots. The standard mean differences (SMD) with 95% confidence interval (95%CI) of ARR and EDSS, mean post-treatment ARR as well as the prevalence of freedom from relapse and EDSS progression were calculated.
The initial search yield 398 articles. Forty articles underwent full-text review. Twelves cohorts and case series (259 MOGAD patients) were included in the meta-analysis. The SMD of pre- and post-treatment ARR was - 0.629 (95% CI -0.874 to -0.384, p <0.001) for all MOGAD patients, -0.440 (95% CI -0.648 to -0.231, p <0.001) in adults, and -0.957 (95% CI -1.450 to -465, p <0.001) in pediatric population. Post-treatment ARR were 0.893 (95% CI 0.174 to 1.612) in adults, and 0.468 (95% CI 0.181 to 0.754) in children. The prevalence of freedom-from-relapse were 68.8% (95% CI 40.2% to 87.8%) and 59.7% (95%CI 37.0% to 78.8%) in adult and pediatric subgroups, respectively. Although EDSS was not significantly changed, 82.7% (95%CI 63.0% to 93.1%) of MOGAD patients had no increasing in EDSS after RTX therapy
This meta-analysis of the cohorts and case series demonstrated the effects of RTX in adult and pediatric MOGAD patients. Although RTX reduced the relapse rate significantly, a large portion of patients continued to relapse. There was substantial heterogeneity in treatment regimens among studies. Prospective controlled studies are needed to validate these results as well as investigate the safety and tolerability of this treatment.