A 28-year-old man presented with acute urine retention. Magnetic resonance imaging (MRI) showed an irregular prostate. Serum prostate specific antigen (PSA) was 1.2ng/mL. Retropubic prostatectomy was performed; histological examination revealed a prostatic malignant phyllodes tumor. This report describes the clinical features and histopathological findings of malignant phyllodes tumor of the prostate and reviews current related literature.