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

• Published in: Neurological sciences Vol. 42; no. 3; pp. 847 - 855
• Main Authors: Nan, Di, Zhang, Ying, Han, Jinming, Jin, Tao
• Format: Journal Article
• Language: English
• Published: Cham Springer International Publishing 01.03.2021; Springer Nature B.V
• Subjects: Adolescent; Adult; Anti-N-Methyl-D-Aspartate Receptor Encephalitis - complications; Anti-N-Methyl-D-Aspartate Receptor Encephalitis - drug therapy; Antibodies; Autoantibodies; Azathioprine; Case reports; Central nervous system; Cerebrospinal fluid; Child; Child, Preschool; Cognitive ability; Cyclophosphamide; Demyelination; Encephalitis; Encephalomyelitis; Glutamic acid receptors; Glutamic acid receptors (ionotropic); Glycoproteins; Humans; Immunotherapy; Intravenous administration; Literature reviews; Medicin och hälsovetenskap; Medicine; Medicine & Public Health; Methylprednisolone; Middle Aged; Monoclonal antibodies; Mycophenolate mofetil; Mycophenolic acid; Myelin; Myelin-Oligodendrocyte Glycoprotein; N-Methyl-D-aspartic acid receptors; Neoplasm Recurrence, Local; Neurology; Neuroradiology; Neurosciences; Neurosurgery; Oligodendrocyte-myelin glycoprotein; Patients; Psychiatry; Review Article; Rituximab; Seizures; Temozolomide; Young Adult; β-Interferon; Systematic review; Anti-NMDA receptor encephalitis; MOG antibody–associated encephalomyelitis; Antibody; Symptoms
• ISSN: 1590-1874; 1590-3478; 1590-3478
• DOI: 10.1007/s10072-020-04942-0

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.