Scrotoschisis is a rare congenital defect of the scrotal sac associated with extrusion of one or both testicles. Only 9 cases have been reported in the literature. The exact mechanism causing this rare congenital defect is unknown. We describe a case of unilateral scrotoschisis in a 35-week preterm infant delivered by cesarean section. The defect was repaired under local anesthesia within a few hours after birth, with good healing noted on follow-up. Scrotoschisis in an infant after delivery by cesarean section has been reported only once before. Although a laceration of scrotum during cesarean section was considered, it seemed unlikely.