• Qingxi Fu, Guangying Wang, Fengyuan Che, Dong Li, and Shougang Wang.
• Department of Neurology, Linyi People's Hospital, Linyi, China.
• Medicine (Baltimore). 2023 Nov 10; 102 (45): e35948e35948.

RationaleFLAIR-hyperintense lesions in anti-myelin oligodendrocyte glycoprotein (MOG)-associated encephalitis with seizures (FLAMES) is a rare clinical phenotype of anti-MOG; immunoglobulin G-associated disease is often misdiagnosed as viral encephalitis in the early stages. Anti-N-methyl-D-aspartate receptor (NMDAR) encephalitis is an autoimmune encephalitis caused by antibodies targeting the GluN1 subunit of the NMDAR. The coexistence of anti-NMDAR encephalitis and FLAMES is very rare.Patient ConcernsA 20-year-old female patient initially presented with seizures accompanied by daytime sleepiness.DiagnosesMagnetic resonance imaging revealed FLAIR-hyperintense lesions in unilateral cerebral cortex. NMDAR antibodies was positive in the cerebrospinal fluid and MOG antibodies in the serum.InterventionsSteroid therapy was administrated.OutcomesThe symptoms completely relieved. At 6-month follow-up, the patient's condition remained stable. Magnetic resonance imaging showed no abnormalities in the unilateral cerebral cortex.ConclusionWhen a patient with anti-NMDAR encephalitis or FLAMES is encountered in clinical practice, the coexistence of these diseases with double-positive anti-NMDAR and MOG antibodies should be considered and adopt appropriate evaluation and treatment.Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc.

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