The unexpected conundrum of endometrioid carcinoma in deep rectal endometriosis arising 11 years after total hysterectomy bilateral salpingo-oophorectomy

Dear Editor: Endometriosis largely affects women, particularly during the fertile age, causing dysmenorrhea, dyspareunia, chronic pelvic pain, and infertility in about 30 % of patients [1]. Its occurrence is estimated to be about 10 % in women in reproductive age, while postmenopausal endometriosis is much less frequent, affecting up to 2–4 % of women, usually in patients with a history of premenopausal disease. Pelvic deep endometriosis infiltrating the rectum and surrounding tissues usually causes severe symptoms, especially pain and obstruction, and its treatment is challenging, because of risk of injury of the pelvic organs and because of the high recurrence rate. Furthermore, even if endometriosis is a benign condition, it has the potential for malignant transformation [2], which occurs in 0.7–1 %, particularly in the ovaries [3]. The endometrial heterotopic tissue is functionally capable of responding to exogenous, endogenous, or local hormonal stimuli, and this estrogen dependence is considered central to its development and progression. For this reason, endometriosis is widely viewed as a disease of the premenopausal age, which normally regresses after menopause because of the lower production of estrogens, even if rare occurrence of endometriosis has been reported in postmenopausal period. In that case, the adipose tissue is supposed to become the main site of endogenous estrogen production even if unopposed hormonal replacement therapy is usually prescribed. However, the onset of deep endometriosis in postmenopausal women submitted to hysterectomy and bilateral oophorectomy for fibromatosis and without hormonal therapy is very rare and represents a conundrum with difficult interpretation. A 54-year-old woman was admitted to our hospital with severe pelvic and anal pain, constipation, and oliguria. Her body mass index was 29. Her personal history included menarche at age 12, with irregular menstrual cycles and dysmenorrhea. She had been pregnant three times, with two spontaneous abortions and one successful pregnancy with cesarean delivery. At the age of 43, she underwent laparotomic hysterectomy and bilateral salpingo-oophorectomy for leiomyomatosis of the uterus. Hormone replacement therapy was not prescribed after surgery. Histologic examination of the resected specimen showed multiple intramural leiomyoma of the uterus, minute borderline cystadenoma, and cyst of the right ovary. At the time of hospitalization, she denied any history of urinary symptoms, and rectal digital examination revealed a palpable, painful, hard, and fixed mass of the pelvis infiltrating the rectal wall. The serum concentrations of carbohydrate antigen 125 (CA 125) and carbohydrate antigen 15.3 (CA 15.3) were 134.4 and 44 .3 U/ml , respec t ive ly. Carcinoembryonic antigen (CEA) and alpha-fetoprotein (AFP) levels were normal. Endoscopic examination revealed the presence of a pelvic mass compressing the walls of the rectum without mucosa infiltration at 8 cm from the anal verge and a large (>5 cm) villous polypoid lesion of the posterior wall of the rectum. Histology on the rectal biopsies from the polyp documented a villous V. Andriola :M. Battaglia : P. Ditonno :M. G. Fiore :M. De Fazio : R. Memeo :D. F. Altomare Department of Emergency and Organ Transplantation, University BAldo Moro^ of Bari, Bari, Italy