Terminal ileum perforation: a rare complication of intestinal endometriosis

Endometriosis is a common benign disease affecting about 10% to 20% of women.1 The frequency of intestinal endometriosis varies between 3% and 37% in women with proven endometriosis. The most often involved site of the disease is the rectosigmoid, followed by the rectovaginal septum, distal ileum, cecum, and the appendix.2 Perforation rarely happens in patients with intestinal endometriosis. Here, we report 2 cases of terminal ileum perforation owing to endometriosis. CASE REPORT Case 1 A 36-year-old woman (gravida 0, para 0) complaining of acute abdominal pain and fever was admitted to our hospital on May 1998. She had a past history of pelvic endometriosis for more than 10 years without regular therapy. On admission, physical examination showed a body temperature of 38°C; muscular tension and rebound tenderness of the abdomen, which was severe in the right lower quadrant; and diminished bowel sound. Thus, we performed abdominal centesis, and found that the abdominal fluid was yellow and thick with total cell concentration of 12/μl. The Rivalta test was positive. The white blood cell count was 10.9 ×109/L. Abdominal X-ray showed dilated bowel with air fluid levels. B-ultrasonography showed free fluid in the right lower quadrant. The gynecological examination was unsatisfied. The patient was diagnosed as having acute abdomen, and then emergent laparotomy was taken. During the operation, we collected a total of 200 ml ascites containing blood and pus, and found that the appendix became swollen and filled with pus. A 5-cm mass was seen at the root of the appendix, protruding into the ileum with a bowel perforation. Therefore, we performed right colectomy. Histological examination after the operation found endometriotic lesions in the muscular layer of the ileum and the appendix serosa. The postoperative course was eventful. The patient was lost to follow-up after being discharged. Case 2 A 43-year-old woman complaining of intermittent abdominal colic and vomiting for 20 days was admitted to our hospital on December 1998. Two years before the admission, the patient had undergone a total abdominal hysterectomy and left ovarian cystectomy because of left ovarian endometriosis and serous endometrial carcinoma, and had received tamoxifen and medroxyprogesterone for six months after the surgery. On May 1997, the patient received a gynecological examination, and the results were negative. On admission, abdominal roentgenography showed dilated bowel with air fluid levels. Thus the patient was diagnosed as having intestinal obstruction, and received emergent laparotomy. During the operation, we found that the small bowel was highly dilated and swollen; the terminal 10-cm ileum was distorted, shortened and rigid. The length of the whole lesion was about 15 cm. A perforation with a flow of chocolate-like fluid was found on the wall of the ileum. Therefore, segmental resection of the ileum with end-to-end anastomosis was performed. The histological examination after the operation showed endometriotic lesions in the ileum muscular and mucosa layers. The postoperative course was eventful. The patient was lost to follow-up after being discharged. DISCUSSION Endometriosis usually involves the pelvic peritoneum, ovaries, cul-de-sac, sacrouterine ligaments, fallopian tubes, rectovaginal septum, rectosigmoid, vulva, vagina, umbilicus, and the small intestine. The scars after caesarean section and episiotomy, and the ureter and bladder may also be affected. Pleura and lung endometriosis are rare. Typically, intestinal endometriosis involves areas where the peritoneum is irregularly folded, such as the rectovaginal septum, rectum, and sigmoid colon. Of the patients with intestinal involvement, 7% had distal ileal involvement, 3.6% had cecal involvement, and 3.0% had involvement of the appendix.1,2 Superficial intestinal endometriosis is often characterized by visible serosal implants. These lesions may be asymptomatic or cause nonspecific cyclical spastic pain. The patients may have chronic constipation, meteorism, cyclical pelvic pain, or colic-like abdominal pain. The cyclical hemorrhage into the serosal foci induces local chronic inflammatory reactions and lesions in the muscle layer of the bowel wall consequently lead to bowel obstruction and adhesions to various adjacent pelvic organs. Intestinal endometriosis may be found in all the layers of the bowel wall, of which the subserosa and the muscularis propria are the most often involved. Intestinal endometriosis is difficult to diagnose because of the lack of characteristic symptom. We recommend that intestinal endometriosis should be considered when a reproductive woman, who had a history of pelvic endometriosis, developed a cyclical bowel discomfort. Ultrasonography and CT scan are not sensitive enough to diagnose the disease. MRI and transanal endosonography is helpful in diagnosis. On histological examination, biopsy of the mucosa is not enough because the typical lesions in the muscularis propria may be missed. Diagnostic laparotomy is suggested in case of surgical emergency. Resection of the involved small bowel segment remains the treatment of choice for patients with intestinal endometriosis since the effect of GnRH analogs and progestins is limited. For asymptomatic superficial intestinal endometriosis, some authors suggested proper follow-up without any treatment. In cases of symptomatic endometriosis, complete resection of the lesion is advocated.3–5 Spontaneous perforation of intestinal endometriosis is extremely rare. To our knowledge, only 3 cases have been reported so far.1,6 In our cases, both of the patients had a history of pelvic endometriosis. Thus we recommend that intestinal endometriosis with perforation should be suspected when a reproductive aged woman with history of endometriosis presents acute abdominal pain. And pelvic organs should be explored during a laparotomy. Moreover, the patients should be closely followed up after the operation.