The ectopic testis is frequently misdiagnosed as cryptorchidism or anorchism. We present a case of three month old male infant diagnosed as right ectopic perineal testis and right hemiscrotal hypoplasia. Ectopic testes are thought to be greater at risk of trauma, testicular torsion, subfertility and malignancy. Although definitive evidence in support of these fears in case of perineal ectopia is lacking due to its rarity and lack of long follow-up. Surgical correction of undescended testes is generally done at about 6 months of age to allow for spontaneous descent. On the other hand there is no need to delay surgery in ectopic testis because possible descent as seen in undescended testis will not occur. Yet the timing of surgery can be individualized for perineal ectopia without any unnecessary delay.