Testis leiomyoma is a very rare neoplasm, it is very difficult to perform a
n exact preoperative diagnosis; only histological examination can prove the
presence of a leiomyoma. In our case radical orchidectomy was performed be
cause of complete substitution of normal parenchyma, the extreme rarity at
long-term follow-up of a reported benign intratesticular neoplasm, and the
imperfect sensitivity of the extemporary histological examination.