Cerebral cortical malformations accompany epilepsy and refractory epilepsy, in particular. However, these malformations may also go together with other neurological, psychiatric and developmental disorders. The presentation of a case who has migraine aims to stress that if there is any coincidental association between cortical dysplasia and migraine, or migraine could be casued by cortical dysplasia. A 14-year-old male patient presented with weekly migrainous headaches that had been continuing for two years. Physical/neurological examination, level of intelligence, school performance, laboratory analyses and routine EEG were all normal. Headache period and sleep-deprived EEGs revealed anterior asymmetric slowing of background; no epileptiform activity. Cerebral magnetic resonance imaging of the case showed dysplastic area in the right frontal, digitation in inter-hemispheric sulcus anterior (cortical dysplasia). It was found that the case had been receiving β blocker treatment for 15 months, to which he responded (mild migraine attack bimonthly). Neuro-diagnostic examination of migraine patients for possible structural lesions might ensure making sense of conditions that can arise later, as well as treating surgically the lesions identified in cases resistant to medical treatment. ©2005, Fırat Üniversitesi, Tıp Fakültesi