Schizencephaly is an uncommon disorder of neuronal migrational characterized by a cerebrospinal fluid–filled cleft, which is lined by gray matter. The cleft extends across the entire cerebral hemisphere, from the ventricular surface (ependyma) to the periphery (pial surface) of the brain. This is a case of open lip schizencephaly diagnosed on MRI with characteristic MR appearance. Note the absent septum pellucidum.

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