Abstract
INTRODUCTION: Interstitial cystitis with Hunner lesion (IC/HL) is an enigmatic, chronic inflammatory disease of the urinary bladder. Few documented cases have reported the IC/HL involving the upper urinary tract. CASE PRESENTATION: A 51-year-old Japanese woman with IC/HL developed bilateral ureteral stenosis and associated hydronephrosis, resulting in increased serum creatinine concentrations. Cystography showed no evidence of bilateral vesicoureteral reflux or bladder deformity. Ureteroscopy showed severe ureteral stenosis due to mucosal hyperplasia at the mid/upper levels of both ureters, leading to the insertion of bilateral ureteral stents. Histological examination of the ureteral lesions showed similar chronic inflammatory changes to the bladder lesions, which were compatible with IC/HL. Bilateral ureteral stenosis persisted after a two-year corticosteroid treatment (prednisolone, 7.5 mg/day), while IC/HL symptoms have disappeared. CONCLUSION: IC/HL could involve the upper ureters, and thus regular follow-up imaging of upper urinary tracts in patients with IC/HL is warranted.