Schizencephaly is a cerebral malformation characterized by a cleft lined with gray matter crossing the entire hemisphere, from the lateral ventricle to the outer surface of the cortex; it would be due to a defect in cell migration during cerebral embryogenesis. Its association with epilepsy is frequent, as well as motor and language disorders and mental retardation. We report the case of a 3-year-old girl who had two open schizencephalic clefts which were objectified on cerebral computed tomography. She also had behavioral and mood disorders, probably related to temporal lobe seizures, which were much improved by sodium valproate.