A case of hydronephrosis with a rare underlying cause in a 35 years old male is described. He reported with pain in the left lumbar region with a past history of left ureterolithotomy. The ultrasound and IVU studies were suggestive of left hydronephroureter. CT Scan showed left hydronephroureter with narrowing at the lower end of left ureter. Ureterorenoscopy (URS) confirmed polypoidal lesions in the left lower ureter, completely obliterating the lumen and involving the whole circumferential wall of the lower ureter. The biopsy of the lesion revealed an inflammatory polyp. Accordingly open surgical intervention was planned. Excision of the lower third of left ureter with ureteric reimplantation was done with a Boari flap. The histopathology report of the lower third of ureter confirmed inverted papilloma of ureter. The patient made a smooth postoperative recovery.
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