Unilateral cerebral cortical encephalitis (UCCE) with myelin oligodendrocyte glycoprotein (MOG)-antibody comprises a new spectrum of disease entities generally presenting seizures. Here, we report a case of a young adult with anti-MOG antibody-associated UCCE who only presented persistent left pulsatile headache. Neurological examination revealed no deficits. Brain MRI showed a fluid-attenuated inversion recovery hyperintense lesion along the swollen left cerebral cortex. The patient was positive for anti-MOG antibodies. We diagnosed him with anti-MOG antibody-associated UCCE. Immediately after the administration of high-dose IV methylprednisolone, the headache diminished. Anti-MOG antibody-associated UCCE is a new differential diagnosis in patients with unilateral chronic pulsatile headache.
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