Primary paratesticular osteosarcoma is an extremely rare malignancy and, to date, only a few cases have been reported. To our knowledge, no such case arising directly from paratesticular soft tissue has been described. We describe a 52-year-old man who presented with left scrotal swelling and an excisional biopsy that revealed a sarcomatous lesion. Left inguinal orchiectomy was performed and histologic examination of the extensively sampled lesion revealed a pure paratesticular osteosarcoma. The patient died 6 months after the diagnosis with disseminated disease.

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