Precursor lesions of papillary urothelial neoplasms have not been well characterized. We reviewed the surgical pathology files of the Johns Hopkins Hospital and three regional hospitals from 1992 to present. Sixteen cases of papillary hyperplasia, defined as undulating urothelium arranged into thin mucosal papillary folds, were identified (in 11 men and five women: age range, 40-89 years). Relative to the diagnosis of papillary hyperplasia, nine patients had a history of papillary urothelial neoplasms; in one of these cases, the patient also had subsequent papillary urothelial neoplasms, and two of these patients had concurrent papillary urothelial neoplasms with papillary hyperplasia. In one of these nine cases, papillary hyperplasia arose in the scar of a prior papillary urothelial neoplasm. In two cases, the patients had concurrent, yet no prior history, of papillary urothelial neoplasms. Of these 11 cases, three had multiple resections showing papillary hyperplasia over time. In case 12, the patient had a history of moderate urothelial atypia. The remaining four patients had no history of papillary urothelial neoplasms or urothelial atypia. We describe papillary hyperplasia as a well-defined entity that is usually asymptomatic and generally found on routine follow-up cystoscopy for papillary urothelial neoplasms. Papillary hyperplasia appears to be a precursor lesion of low-grade papillary urothelial neoplasms.