Primary leiomyosarcoma of the prostate is extremely rare. It accounts for less than 0.1% of prostate neoplasms and has a poor prognosis. Most patients are presented with obstructive symptoms, perineal pain, burning on ejaculation, and weight loss. Multimodality treatment combinations including surgery like prostatectomy or cystoprostatectomy, preopreoperative or postoperative radiation therapy, and neoadjuvant or adjuvant chemotherapy have been used in the management of leiomyosarcomas of the prostate, but there are no standard treatment recommendations. Herein, we present a 62-year-old male suffering from recurrent hematuria and rapidly progressive prostatism, diagnosed as leiomyosarcoma of the prostate.