Background: Small cell carcinoma (SCC) of the prostate represents a rare form of prostatic carcinoma. While the tumor is often considered to arise from neuroendocrine proliferation or possibly dedifferentiation of an acinar carcinoma, the precise etiology remains uncertain. The diagnosis of prostatic SCC in urine has to date not been described. Methods: A retrospective review was performed at a tertiary-care hospital, and 3 patients with prostatic SCC in voided urinary specimens were identified. The following clinical data were collected for each case: age, gender, treatment and follow-up information, when available. Results: The patient age range was 70-86 years, all male. Two patients had known metastatic adenocarcinoma of the prostate, and 1 had recently presented with prostatic SCC. One patient with metastatic disease died shortly after diagnosis, the other was lost to follow-up. The third patient with a recent presentation has yet to have a treatment plan finalized. Conclusions: Our results highlight the importance of making this uncommon diagnosis as it may carry significant treatment and prognostic importance. Future work should hopefully clarify the role of ERG gene rearrangements in the pathogenesis of prostatic SCC, as there is a potential role here for targeted therapy.