Schizencephaly is a rare disorder of cerebral cortical development with a frequency of 1.54 / 100,000. It is caused by defective migration of neurons with the subsequent formation of fissures extending from the pia mater of the cerebral wall to the underlying ventricular surface. It is often associated with other brain abnormalities such as polymicrogyria, pachygyria, ventriculomegaly, heterotopia of the gray matter, agenesis of the septum pellucidum, agenesis of the corpus callosum and others. It is most often accompanied by mental retardation, learning difficulties and epileptic seizures.An atypical case of late epileptic onset in a 21-year-old man with normal neuropsychiatric development due to schizencephaly and agenesis of the septum pellucidum is presented.
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