Prostate sarcoma is an extremely rare malignancy that accounts for only %0.1 of all neoplasms of the prostate gland. Primary prostate leiomyosarcoma (PLSOP) is the most common subtype in adults. Due to the fact that it is an extremely rare malignancy, case reports have been reported frequently and several publications in the form of case series. The number of case reports in the world is less than 200. Our opinion is that publishing such rare diseases and bringing them to the literature will have positive benefits both scientifically and for the patients. We present a patient with PLSOP and discuss the clinical, diagnostic and therapeutic aspects of this rare malignancy. Keywords: Prostate, Leiomyosarcoma, Cancer, Prognosis.