Perineal testis is a very rare congenital anomaly. Since the first case of the disease reported by John Hunter in 1786, more than 113 cases have been reported in the English literature, and 13 cases have been reported in the Japanese literature. The 14th and 15th cases are presented herein. Case 1 was a 38-year-old man who came to our clinic with the chief complaint of discomfort of the perineum. Physical examination revealed emptiness of the left scrotal contents and there was a small hen's-egg growth mass corresponding to the normal testis at the left side of the perineum. Excretory urogram showed no abnormality. Orchiopexy to the left scrotum was performed. On surgical exploration the gubernaculum testis was found obviously fixed to the perineum. Histologic examination of the biopsied ectopic testis revealed mild hypospermatogenesis. Case 2 was a 2-year-old boy who was referred to our clinic with the chief complaint of emptiness of the right scrotal contents. Physical examination revealed a bean-sized mass corresponding to the normal testis at the right side of the perineum. Orchiopexy to the right scrotum was performed. On surgical exploration the gubernaculum testis was found to be fixed to the perineum
