• Hongbing Nie, Haifeng Gao, Yongqiu Li, and Yaoyao Shen.
• Department of Neurology, Jiangxi Provincial People's Hospital Affiliated to Nanchang University, Nanchang, Jiangxi Province, China.
• Medicine (Baltimore). 2021 May 21; 100 (20): e26087.

RationaleNowadays, myelin oligodendrocyte glycoprotein (MOG)-antibody-associated disease (MOGAD) is regarded as an independent inflammatory demyelinating disease. Here, we report a rare case of unilateral cerebral cortical encephalitis (CCE) with positive anti-MOG antibodies.Patient ConcernsA 19-year-old woman was admitted to our hospital owing to acute onset fever and headache. Four days later, she experienced a focal seizure that progressed to generalized tonic-clonic seizures.DiagnosisBrain magnetic resonance imaging (MRI) demonstrated cortical lesions in the left cerebral hemisphere on T2-weighted fluid-attenuated inversion recovery imaging. The patient was positive for anti-MOG antibodies in serum and diagnosed with anti-MOG antibody-associated unilateral CCE.InterventionsShe was administrated with intravenous methylprednisolone followed by oral corticosteroids.OutcomesOn day 14 after admission, a repeat MRI revealed partial resolution of the initial abnormalities. The patient received a quick recovery without residual symptoms.ConclusionsUnilateral CCE with positive anti-MOG antibodies has emerged as a special clinical phenotype of MOGAD. It should be emphasized that the characteristic neuroradiological features of CCE would be an important clue to the correct diagnosis of MOGAD.Copyright © 2021 the Author(s). Published by Wolters Kluwer Health, Inc.

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