Perineal ectopic testis is a rare congenital anomaly in which the testis is abnormally situated between the penoscrotal raphe and the genitofemoral fold. Etiopathogenesis of perineal ectopic testis is controversial. Perineal ectopic testis is frequently associated an inguinal hernia and can sometimes be associated with other disorder such as hypospadias and scrotal anomalies. Perineal ectopic testis is relatively rare but is easily recognized and treated by orchiopexy. Diagnosis is easily based on physical examination that reveals an empty scrotum and a soft tissue mass in perineal region. Histopathologically, the course of perineal ectopic testis is better than undescended testis. We report a 21 years old male with perineal pain and empty right scrotal sac. He was diagnosed to have perineal ectopic testis and was treated with orchiopexy.