Background Anti-N-methyl-D-aspartate receptor (anti-NMDAR) encephalitis is an autoimmune disease of the central nervous system (CNS) that often causes a range of neuropsychiatric symptoms; but with a relatively low recurrence rate. Anti-NMDAR encephalitis may occur with anti-myelin oligodendrocyte glycoprotein (MOG) encephalitis simultaneously or followed by anti-MOG encephalitis after several months. Case presentation We presented a case of recurrent anti-NMDAR encephalitis followed by anti-MOG encephalitis. A 39-year-old man was admitted to the hospital with five episodes of convulsive seizures, psychoses and memory deficits. With a positive Anti-NMDAR antibody in the cerebrospinal fluid (CSF), after being treated with intravenous immunoglobulins (IVIG) 22.5 g/day for 7 days, intravenous methylprednisolone pulse (IVMP) 500 mg/day for 4 days, and followed by IVMP 250 mg/day for 3 days, the patient was recovered completely. After 1 month, the patient developed disturbances of consciousness, speech difficulties and convulsions. With the positive result of Anti-NMDAR antibodies in the CSF again, we confirmed the diagnosis of recurrent anti-NMDAR encephalitis. The patient was then fully recovered after treatment with IVIG, IVMP and rituximab. Six months later, the patient was readmitted to the hospital with occasional convulsions. With a positive serum MOG antibody and a lesion in the right brainstem by brain magnetic resonance imaging (MRI), the patient was confirmed for anti-MOG encephalitis. The patient was fully recovered again after treatment with IVMP and rituximab, with no neurological sequels remained. Conclusion Patients with positive MOG antibodies are more likely to have brainstem MRI lesions than those with anti-NMDAR encephalitis. Rituximab is recommended for recurrent anti-NMDAR and anti-MOG encephalitis.
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