Background: Myelin oligodendrocyte glycoprotein (MOG) antibody positive utilateral cerebral cortical encephalitis (UCCE) comprises a new entity with heterogeneity which usually present as epilepsy. Cases with cephalalgia as the only clinical feature were rare reported. Here, We report a case with MOG antibody positive UCCE who only presented with refractory cephalalgia and had a good response to glucocorticoid. MOG antibody-related UCCE should be identified when patients present with refractory

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... headache and it may represent benign cortical encephalitis.Case Presentation: The case of a 41-year-old woman with MOG antibody positive UCCE presented with refractory cephalalgia. Neurological examination is normal, and fundus examination suggested bilateral optic nerve head edema. Brain MRI discovered Flair hyperintense lesions in the sulci of right temporoparietal occipital cortex. MOG antibody was positive both in the serum and cerebrospinal fluid. When the patient was treated with intravenous methylprednisolone, her headache completely disappeared and brain MRI showed partly recovery. At the follow-up of 6 months after discharge, she had no relapse, serum MOG antibody was negative and the high signal intensity on Flair had completely disappeared.Conclusions: MOG antibody-positive UCCE with headache as the single clinical symptom may represent benign cortical encephalitis.