Primary testicular leiomyosarcomas (LMS) are very rare tumours, with only 20 cases reported so far in the literature (Abdullazade et al. Turk Patoloji Derg 29:227-230, 2013; Komeya et al. Indian J Urol 28:343-346, 2012). We present a case of a 70-year-old male patient who presented with recurrence in the right hemiscrotum 10 weeks after right inguinal orchidectomy for a testicular mass which diagnosed as primary intratesticular leiomyosarcoma. He was treated with wide local excision and adjuvant radiotherapy.

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