Müllerianosis of the urinary tract is a very rare and morphologically complex tumor-like lesion. It is composed of several types of müllerian-type lesions, including endometriosis, endocervicosis, and endosalpingiosis. We present a case of ureteral müllerianosis in woman with a history of a cesarean section about 20 years previously but with silent clinical symptoms. She was treated with segmental ureteral resection, followed by ureteroureteral anastomosis. The pathogenesis and treatment of this lesion are discussed.