Squamous papilloma of the renal pelvis is an extremely rare entity. To the best of our knowledge, no case has been reported till date. A 45-year-old female presented to us with flank pain for two months. She had undergone left-sided percutaneous nephrolithotomy three years ago. Preoperative evaluation suggested the presence of a transitional cell tumor arising from the left renal pelvis. Diethylene-triamine-pentaacetic acid renogram showed a non-functional left kidney. She underwent left nephroureterectomy. Histopathology showed squamous papilloma of the renal pelvis. Subsequent follow-ups have been uneventful.