Endometriosis outside the pelvis is rare and most cases occur in surgical scars after operations involving the female genital tract. Fistulae involving the uterus are also very rare, usually being the result of postpartum and postoperative complications. In the present report, a case of a 44-year-old patient with an endometriotic uterocutaneous fistula is described. The patient presented 6 years after her fourth cesarean section with a painful nodule on the cesarean scar, which was bleeding during menstruation. The lesion extended to the uterine fundus, connecting the endometrial cavity with the skin. This is merely the second case of a uterocutaneous fistula to be reported in the literature and the first case developed on grounds of endometriosis.