Introduction : Leiomyosarcoma (LMS) of the prostate is an extremely rare and aggressive tumor that presents with nonspecific signs and symptoms. Treatment guidelines are not yet established. Case Presentation : We report two cases of LMS of the prostate. The presenting symptom was hematuria, and diagnosis was ascertained through a transurethral resection of the prostate for the 2 patients. The treatment course consisted of three courses of chemotherapy with gemcitabine and docetaxel, radical prostatectomy, and postoperative radiation therapy for the first patient and three courses of gemcitabine and radiation therapy of the prostate and the whole pelvis for the second patient. The follow-up of our 2 patients was 9 and 12 months, respectively. Recurrence occurred 10 months after treatment completion for the second case. No recurrence was noticed in the first case. Conclusion : These two cases highlight the importance of a multimodal approach to yield the best outcomes.