攝護腺平滑肌肉瘤（leiomyosarcoma）十分罕見，至目前只有少於一百例的病例被報告。在早期的報告中，平滑肌肉瘤被認為多發生在小孩和成人身上，但近期的報告指出，攝護腺平滑肌肉瘤只發生在成年人。由於大家的經驗都不多，至今仍沒有統一的治療流程，但一般較同意早期診斷和積極治療對癒後有正面意義。本院報告一位27歲男性病人，從診斷及手術治療至今已四年，門診定期追蹤並無復發的現象。我們同時對此種罕見惡性腫瘤的相關文獻進行回顧。

Leiomyosarcoma of the prostate is a rare neoplasm;approximately less than 100 cases have been documented. In early studies, leiomyosarcoma was described in children and adults, but most current series suggest that prostate leiomyosarcoma only occurs in adults. Since this neoplasm is an infrequent lesion, treatment has not been well established. Most authors advise early diagnosis and emphasize the importance of radical surgery for localized disease. We report herein one case of prostatic leiomyosarcoma, in which a 27-yr-old man is still alive with no evidence of metastases 4 years post-operatively. Relevant literature of this uncommon and aggressive malignancy is also reviewed.