Leiomyomata of the urinary and male genital tract are extremely rare. They have been reported throughout the genitourinary male tract and the most common localization is the renal capsule. However, leiomyomas of the epididimis, spermatic cord, tunica albuginea and testis have been reported. We report diagnostic confirmation of a paratesticular leiomyoma in an azoospermic patient undergoing a testicular sperm extraction for intracytoplasmic sperm injection (TESE/ICSI) procedure.