Patient. A 65‐year old man who had previously undergone surgery for benign prostatic hyperplasia presented with symptoms of recurrent bladder outflow obstruction. Cystoscopy revealed the presence of recurrent tissue.Results. Histology identified a leiomyosarcoma. Several years after further surgery and radiotherapy, the patient presented with secondaries in the lungs and brain, but there was no evidence of local recurrence.Discussion. It is suggested that combined modality therapy (conservative surgical resection followed by adjuvant radical radiotherapy) be the initial treatment for prostatic leiomyosarcoma.