Schizencephaly is an uncommon congenital malformation of the central
nervous system which affects the development of the cerebral cortex. It is
defined as the gray matter lined filled cleft with cerebrospinal fluid that
extends from the pial surface to the ventricle. There exists a paucity in
scientific literature regarding the association between schizencephaly and
psychosis. We report a case of a 36-year-old male, presented with worsening
crisis of aggression, disorganized behavior and auditory hallucinations over
20 years. CT brain scan revealed unilateral left open lip schizencephaly, a
finding during his recent admission to the psychiatry ward.