We report here a case of bilateral pyeloureteritis cystica. A 67-year-old woman was admitted to our hospital with asymptomatic macrohematuria in September 1999. Drip infusion pyelography and enhanced computed tomography demonstrated multiple small, round filling defects in both renal pelvises and ureters. Ureteroscopy and cold punch biopsy were performed, and histological examination revealed pyeloureteritis cystica. This patient was not given adjuvant therapy but was carefully followed up for 3 years and 6 months postoperatively.