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Clinical features and management of coexisting anti-N-methyl-D-aspartate receptor encephalitis and myelin oligodendrocyte glycoprotein antibody–associated encephalomyelitis: a case report and review of the literature

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Abstract

Background

Anti-N-methyl-D-aspartate (NMDA) receptor encephalitis is an autoimmune encephalitis caused by antibodies targeting the GluN1 subunit of NMDA receptors. Myelin oligodendrocyte glycoprotein (MOG) antibody disorders are now widely accepted as peculiar neuroimmunological diseases with specific clinical and pathological features. Some rare cases of overlapping anti-NMDA receptor encephalitis and MOG antibody–associated diseases have been reported, presenting complex clinical symptoms that make the disease more difficult to recognize.

Method

In accordance with the Preferred Reporting Items for Systematic reviews and Meta-Analyses (PRISMA) guidelines, the terms “NMDAR” and “MOG,” “NMDAR” and “demyelination,” and “MOG” and “encephalitis” were searched in PubMed. Clinical cases with dual-positive anti-NMDA cerebrospinal fluid receptors and MOG serum antibodies during the disease course were included in this study.

Results

A total of 25 patients were analyzed in this study. The age at onset ranged from 3 to 54 years. The median number of relapses was 2.8. Administration of intravenous methylprednisolone and immunoglobulin was the most widely used treatment strategy (19/25 patients). Second-line treatments such as administration of mycophenolate mofetil, rituximab, interferon-β, azathioprine, cyclophosphamide, and temozolomide were also reported, followed by good outcomes.

Conclusions

The rates of coexisting anti-NMDA receptor encephalitis and MOG antibody–associated encephalomyelitis may be underestimated. Clinical symptoms such as seizures and cognitive decline accompanied by atypical central nervous system demyelination serve as warning signs of possible coexisting anti-NMDA receptor encephalitis and MOG antibody–associated encephalomyelitis. These patients could achieve good outcomes under proper immunotherapies.

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Funding

Dr. Jin has received support from the General Program of the National Natural Science Foundation of China (No. 81671177), Natural Science Foundation of Jilin Province Science and Technology Development Plan Project (20190201043JC), Key Research and Development Project of Social Development Division of Jilin Science and Technology Department (20200403109SF), Special Project for Health Professionals of Jilin Provincial Finance Department (JLSWSRCZX2020-0056),and the grants from the Swedish Research Council (No. 2015-03005) and grants from the First hospital, Jilin University of China. The funding body did not play any role in the study design; in the collection, analysis, and interpretation of data; in the writing of the manuscript; or in the decision to submit the manuscript for publication.

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  1. Jinming Han

    Present address: Department of Clinical Neuroscience, Karolinska Institutet, Solna, Sweden

  2. Di Nan and Ying Zhang contributed equally to this work.

Authors and Affiliations

  1. Department of Neurology and Neuroscience Center, the First Hospital of Jilin University, Xinmin Street 1#, Changchun, 130021, China

    Di Nan, Ying Zhang, Jinming Han & Tao Jin

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DN drafted the manuscript. JH and YZ helped to prepare the figures and collected data. JH and TJ performed a critical revision. All authors have read and approved the manuscript.

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Correspondence to Tao Jin.

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Nan, D., Zhang, Y., Han, J. et al. Clinical features and management of coexisting anti-N-methyl-D-aspartate receptor encephalitis and myelin oligodendrocyte glycoprotein antibody–associated encephalomyelitis: a case report and review of the literature. Neurol Sci 42, 847–855 (2021). https://doi.org/10.1007/s10072-020-04942-0

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