Abstract Follicle-stimulating hormone (FSH) is a central hormone in mammalian reproductive biology. The FSH receptor (FSHR), which was previously believed to be expressed primarily in the ovary and testis, was recently found to be expressed in the tumor blood vessels of many solid tumor types, including prostate adenocarcinoma, urothelial carcinoma, and renal cell carcinoma. While the biologic significance of FSHR in tumor blood vessels has yet to be elucidated, FSHR may contribute to neoangiogenesis. FSHR has been reported to be expressed by prostate cancer cells and, thus, targeting FSHR in prostate cancer may be of particular utility. In this report, we discuss the finding of FSHR in tumor blood vessels and review the literature concerning FSHR in genitourinary malignancy. We also discuss the features that make FSHR an appealing target for therapeutic and imaging purposes and the potential utility of FSHR as a prognostic and/or predictive biomarker in genitourinary cancers.