Abstract
Schizencephaly is a rare congenital anomaly characterized by the formation of abnormal clefts in the brain. Despite the association of psychotic symptoms with various brain abnormalities or insults, their occurrence in individuals with schizencephaly is relatively infrequent. The association of bipolar disorder, with or without psychosis, with schizencephaly is rarer. A systematic search on PubMed using "Schizencephaly AND Bipolar Disorder" yielded only four case studies specifically addressing the connection between these two conditions. Here, we present a case of a 22-year-old male patient with a history of childhood seizures who developed first episode psychosis along with manic symptoms and was found to have closed-lip schizencephaly.