We report an exceptionally rare case of ureteral stenosis caused by an extraskeletal chondroma. A woman in her 60s underwent laparoscopic hysterectomy for presumed uterine fibroid torsion, but later developed severe right flank pain. Imaging revealed hydronephrosis and ureteral stricture suspicious for tumor. Ureteroscopy with biopsy confirmed chondroma, representing only the second reported case worldwide after 1973. The patient has been managed with periodic ureteral stent exchanges, leading to symptom relief and improved hydronephrosis. This case highlights the extreme rarity of ureteral chondroma, the diagnostic challenge of differentiating it from malignancy, and the importance of histopathological confirmation.

© 2025 The Authors. Published by Elsevier Inc.