Endometriosis of the urinary tract is infrequent. The ureters and kidneys are the least usual place of localization. Endometriosis of the ureter often leads to silent loss of renal function due to delayed diagnosis. We report a case of a premenopausal female with endometriosis of the left distal ureter, presenting an infection of the urinary tract and having reported previous incidents of menorrhagia and left flank renal pain with automatic recession. Pharmacological treatment was applied with a satisfactory outcome. A short review of the literature is presented.