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Leiomyosarcoma of the prostate-an unexpected histopathological outcome. Prostate leiomyosarcoma is an extremely rare and highly aggressive neoplasm that accounts for >0.1% of all primary prostate malignancies. We report a case of a patient, presenting with recurrent episodes of dysuria, who had been diagnosed and operated for benign prostatic hyperplasia 1 month earlier, and now presented with similar symptoms postoperatively. Trans-rectal biopsy of the prostate was carried out and histopathology revealed leiomyosarcoma of the prostate.