Introduction

In this case report, we describe a patient who had a long history of abdominal complaints. She came to our emergency department (ED) with a rare case of acute bowel obstruction caused by endometriosis. The diagnosis of endometriosis is usually made after years of complaints with the absence of a distinct acute onset [1,2].

Case presentation

A 31-year-old Caucasian woman with increasing abdominal distension, pain and nausea was referred to our department by gastroenterologists at the ED. She had had abdominal pain and chronic constipation symptoms since the age of 17 years. These complaints had been extensively analyzed several years earlier by the gastroenterology department, when no abnormalities were identified in blood tests or a colonoscopy. One month prior to her presentation at the ED, she had been admitted to the surgical department with complaints of decreased intake, nausea and a 10-day absence of defecation. Blood tests did not show abnormalities. An abdominal X-ray showed signs of constipation. After conservative treatment with enemas and oral debulking agents, her complaints decreased and she was dismissed from hospital.

During the intake at the ED, she was found to have no history of weight loss, and, with the use of bisacodyl and close monitoring of her diet, she had managed to regulate her bowel movements, with the exception of her recent admission. For the preceding 1 year, the patient had desired to become pregnant. Therefore, she had stopped using oral contraceptives, after which she had a regular cycle without intermediate bleeding or abnormal vaginal discharge.

In her physical examination, we encountered a painful woman with a normal body temperature. She had abdominal distension, hypertympanic percussion, active bowel movements and tenderness of the abdomen without signs of abdominal guarding. Additional blood tests showed a normal hemoglobin level of 7.8 (normal range, 7 to 10mmol/l), without any signs of an infection with 6.8×104 leukocytes (normal range, 5 to 10×104) and a C-reactive protein level of 8.0 (normal level, <10mg/l). An abdominal X-ray (Figure 1A) and subsequent abdominal computed tomography (CT) (Figure 1B,C) showed a significantly inflated cecum of 9cm and an obstruction in the sigmoid colon with a proximal distension of the colon. Because of the expansion of the cecum, a decision was made to perform emergency colonoscopy for desufflation; however, the obstruction could not be passed with a scope.

Figure 1
figure 1

Pre-operative imaging studies. X-ray (A) and computed tomographic scans (B, C) of the patient’s abdomen show a significantly inflated cecum and an obstruction in the sigmoid colon with a proximal distension of the colon.

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Given the threat of cecal blow-out, it was decided to perform a laparotomy. During the surgical procedure, a solitary stenosis was found in the sigmoid colon that appeared as a malignancy. Therefore, an oncological sigmoid resection with primary anastomosis was performed. Because of the distension of the cecum, a temporary loop ileostomy was constructed. Upon further inspection of the abdomen, no abnormalities were found in the liver, peritoneum or pelvis. The patient recovered quickly after the procedure and could be discharged after 1 week, with the intention to perform a loop ileostomy reversal after 3 months.

A macroscopic pathological examination of the resected sigmoid showed extensive fibrotic foci with induration in the intestinal wall without mucosal abnormalities (Figures 2 and 3). In the sigmoid wall, a tumor 4.4cm in size was found; however, this did not appear to have an obvious relationship to the mucosa. Microscopically, the tumor showed epithelial tubular structures surrounded by what was interpreted as cytogenic stroma and focal hemorrhage remnants, suggestive of endometriosis (Figure 4). Foci of tubes with ciliated columnar epithelium were present in some lymph nodes.

Figure 2
figure 2

Pathological specimen obtained during sigmoid resection.

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Figure 3
figure 3

Extensive fibrotic foci can be seen with induration in the intestinal wall without mucosal abnormalities.

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Figure 4
figure 4

Microscopically, the tumor had epithelial tubular structures surrounded by arguably cytogenetic stroma and focal hemorrhage remnants suggestive of endometriosis.

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Discussion

Endometriosis is defined as the implantation of endometrium outside the uterus [3,4]. In 1927, Sampson theorized that endometriosis results from retrograde deposition of endometrial remains during menstruation [5]. These endometrial cells remain sensitive to gonadotropic hormones and react to hormonal changes during the menstrual cycle. Sampson’s theory does not fully explain why endometriosis occurs only in a select group of women, and to date the exact etiology remains unknown. Recent research has shown that differences in genetic and immunological factors exist between affected and unaffected women [1,6,7].

Endometriosis affects 10% to 12% of fertile women, with the number of cases diagnosed peaking between the ages of 29 to 39 years. First onset can be as early as adolescence, making it prone to being missed as a diagnosis [1-3,8,9]. Endometriosis can occur in the entire abdominal cavity and is often classified into three groups: peritoneal endometriosis, ovarian endometriosis and deep infiltrating endometriosis [10]. Deep infiltrating endometriosis is not clearly defined, but usually it is described as an ingrowth of over 5mm into the peritoneum and underlying structures [1,4,7,10]. In 5% to 12% of the women affected by endometrioses, there is infiltrating growth in the rectosigmoid [2,3,10,11]. Additional common locations for ingrowths are the uterosacral ligaments, vagina, bladder and ureters [10].

Symptoms of deep infiltrating endometriosis of the rectosigmoid include dysmenorrhea, constipation, diarrhea, pain during defecation, dyspareunia and periodic rectal bleeding during menstruation [2,3,10,12,13]. Late presenting symptoms can be chronic because of extensive fibrosis that occurs as a result of the endometriosis depositions. Physical examination often provides insufficient clues to lead to suspicion of deep infiltrating endometriosis; hence, imaging is an important aspect of making the diagnosis [4,7]. To diagnose deep infiltrating endometriosis, transvaginal ultrasound is a highly reliable imaging technique to complement the gold standard of laparoscopy [1,3,4]. Transvaginal ultrasound has a reported sensitivity of 91% and specificity of 98% [14]. For large depositions in the cavum Douglasi, and as pre-operative imaging, a magnetic resonance imaging scan with rectal contrast has additional value and is superior to CT [3,15].

Deep infiltrating endometriosis often presents at a late stage with extensive complaints due to extensive fibrosis caused by chronic inflammation of the endometriosis. Anti-inflammatory and hormonal therapies have little to no effect on the symptoms and therefore provide no added value in this stage of the disease [10]. Surgical intervention has been argued to be the treatment of choice at this stage. In recent years, a number of new techniques have been developed; however, the gold standard remains segmental resection laparoscopy or, if this is complicated because of adhesions, laparotomy [2,11,16]. In this otherwise healthy patient population, fewer complications are seen than with oncological colorectal surgery [17]. The most frequent complications are rectovaginal fistula (2% to 8%) and anastomotic leakage (0 to 3%). Similarly to oncological colorectal surgery, there is a strong link between the risk of anastomotic leakage and the position of the anastomoses [2,10,11,13,17]. There is less need for the construction of loop ileostomy in this group of healthy young women. This decision making should additionally take into consideration the severe psychological effects that accompany a stoma at this age [17]. A partial resection or curettage of the lesion is an alternative to a bowel resection with a lesion smaller than 2cm [2,4,10]. The risk of recurrence after an intervention for infiltrating endometriosis varies widely and is difficult to assess because of differing research methods and follow-up durations. Most review authors state a recurrence risk of 5% to 25% [2,10,11]. Remarkably, this is not influenced by the radicality of the resection [12].

Conclusions

This case illustrates that infiltrating endometriosis is a disabling disease. Emergency room physicians and surgeons should be aware that it can present as an acute obstruction and should be considered in diagnosing women of childbearing age. In our patient, a CT scan, rather than an abdominal X-ray, might have revealed the pending obstruction in an earlier phase. In exceptional cases, colon obstruction by infiltrating endometriosis can require an emergency laparotomy with resection of the affected bowel segment. In less urgent cases, more planning is recommended, along with discussion of the case, including medical images, among a multidisciplinary endometriosis team.

Consent

Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.