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The incidence of urothelial carcinoma (UC; formerly transitional cell carcinoma) is higher among renal transplant recipients compared with the general population. Upper urinary tract UC (UUT-UC) of allograft urothelium is a rare event with approximately 40 cases reported in the literature. Herein, we describe the clinical presentation and management of UUT-UC in a transplant ureter 10 years after deceased donor kidney transplantation.