Abstract This report describes a case of primary osteosarcoma of the testis in a 60-year-old man. Treatment consisted of an inguinal orchiectomy with no adjuvant therapy. The patient is alive and doing well without recurrent disease at 18 months after diagnosis. Only three reports have been published on primary osteosarcoma of the testis. The origin of this tumor from undifferentiated mesenchymal cells or from a malignant transformation of pre-existing teratomatous elements is still unclear. Management guidelines are difficult to establish due to the rarity of such tumors, but inguinal orchiectomy with careful follow up appears to be sufficient treatment.