Urothelial recurrence of renal cell carcinoma after radical surgery is exceedingly rare. We report on a 48-year-old man who underwent upper urinary tract flexible endoscopy to define a filling defect, which was found to be a perforating renal cell carcinoma. After radical nephrectomy the patient had multiple recurrences on the urinary surfaces, ureter and urethra. Urothelial lesions were treated endoscopically. He remains free of tumor 42 months after the last treatment.