A seven-year-old boy with migraine and a family history of hemiplegic migraine is described who, during an exacerbation, developed dysphasia and right hemiparesis. A CT scan showed a hypodense left cerebral lesion. Angiography revealed occlusion of the left middle cerebral artery at its origin. This represents the youngest case of stroke with migraine and, to our knowledge, is the first case report of angiogram-documented middle cerebral occlusion associated with migraine.