Small cell carcinoma of the prostate is rare and is thought to behave clinically as a highly aggressive neoplasm. We report on a patient with pure small cell carcinoma of the prostate presenting as multiple organ metastases. Immunohistochemical studies of the tumor cells revealed positive reactions to chromogranin A, synaptophysin, and cytokeratin stains, and a negative reaction to PSA stain. This patient received 5 courses of gemcitabine/carboplatin chemotherapy after transurethral resection of the prostate, and the previous metastatic bone lesions showed partial resolution.