The case records of 2 patients recently treated at our medical centers with prostatic cystic epithelial-stromal tumor (ages 22 and 62 years), as well as 14 cases previously reported in the literature were reviewed to obtain a consensus as to the therapy for this uncommon malignancy. Patients with prostatic cystic epithelial-stromal tumor often present with obstructive voiding symptoms and a palpable suprapubic mass. Computerized tomography typically reveals a huge, complex retrovesical mass with displacement of surrounding pelvic and abdominal structures, which may invade locally into the bladder, ureters or rectal wall. Our experience with immunohistochemical staining of these tumors suggests an epithelial component that is positive for prostate specific antigen, prostatic acid phosphatase, epithelial membrane antigen, chorioembryonic antigen and cytokeratin, and a stromal component that is positive for vimentin, desmin, cytokeratin and myosin. Rapid recurrences are the rule in patients in whom the tumor is incompletely resected. Histological evidence of malignant transformation and distant metastases has been reported in these neoplasms. An aggressive surgical approach aimed at total removal of this pelvic tumor will be discussed.