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Dyke-Davidoff-Masson syndrome presenting as recurrent chronic headache in the late adult life. Dyke-Davidoff-Masson syndrome (DDMS) (also referred to as cerebral hemiatrophy) is a rare condition characterized by seizures, facial asymmetry, contralateral spastic hemiplegia, or hemiparesis, with or without learning difficulties. It usually presents in the early childhood or late adolescence. The diagnosis is mainly associated with the presence of radiologic findings which include contralateral cerebral hemiatrophy with ipsilateral dilatation of the lateral ventricle and hypertrophy of the sinuses. Here, we report a case of a 49-year-old female patient who presented with chronic headache episodes for 10 years, and radiological findings revealed the DDMS.