Rare manifestation of endometriosis-related intestinal obstruction in the small bowel of a middle-aged female: a case report

Manager Save my selection Surgical Case Report Rare manifestation of endometriosis-related intestinal obstruction in the small bowel of a middle-aged female: a case report Alaswad, Mohammed MDa,*; Haydar, Hasan MDa; Almohamed, Ahmad MDa; Qatza, Ayhama; Alomar, Bauthainaa; Youssef, Simon MD, MScb; Barshiny, Sari Medhat MD, MScc aFaculty of Human Medicine, University of Hama, Hama, Syria bPathology Department, Hama National Hospital, Hama, Syria cAl Mahabah Hospital, Hama, Syria *Corresponding author. Address: Faculty of Medicine, University of Hama, Hama Governorate, Hama 0000, Syria, 00963935699890, E-mail:[email protected] (M. Alaswad). Sponsorships or competing interests that may be relevant to content are disclosed at the end of this article. This is an open access article distributed under the terms of the Creative Commons Attribution-Non Commercial License 4.0 (CCBY-NC), where it is permissible to download, share, remix, transform, and buildup the work provided it is properly cited. The work cannot be used commercially without permission from the journal. Endometriosis, the implantation and proliferation of endometrial tissue outside the uterus, is a chronic inflammatory disease affecting 6–10% of menstruating women. While ovarian involvement is the most common, intestinal endometriosis accounts for 3–37% of cases of women diagnosed with endometriosis, with distal ileal endometriosis (DIE) causing bowel obstruction being particularly rare. Case presentation: A 43-year-old Syrian woman with a history of infertility presented with generalized abdominal pain, vomiting, and fever. Imaging studies revealed dilated intestinal loops and air-fluid levels. Surgical exploration identified an edematous, volvulated segment of the terminal ileum causing small bowel obstruction. Microscopic examination of the resected segment confirmed the diagnosis of ileal endometriosis, showing endometrial glands and stroma within the intestinal wall.

Intestinal endometriosis is typically seen in the rectosigmoid colon but rarely affects the small intestine, with the ileum being an uncommon site. Symptoms of DIE often mimic other gastrointestinal disorders, making the diagnosis difficult. Surgical approach remains the gold standard for confirming the diagnosis and management when obstruction occurs.

This case underscores the importance of considering intestinal endometriosis in the differential diagnosis of chronic or acute abdominal pain and bowel obstruction, especially in women of reproductive age with a history of infertility. Plain Language SummaryEndometriosis, a condition where tissue similar to the lining inside the uterus grows outside it, can sometimes affect the intestines, though this is rare. A 43-year-old woman from Syria experienced severe abdominal pain and vomiting due to a blockage in her small intestine. Surgery revealed that the blockage was caused by endometriosis in the ileum, a part of the small intestine. This case highlights the need for doctors to consider intestinal endometriosis as a possible cause of abdominal pain and bowel obstruction, especially in women of reproductive age who have infertility issues. Text is machine generated and may contain inaccuracies. FAQ

The term “endometriosis” refers to the implantation and proliferation of normal endometrial tissue outside the uterus and myometrium[1-3]. It is a chronic inflammatory disease that affects 6–10% of menstrual women[4]. Endometriosis frequently affects pelvic organs, with the ovaries and fallopian tubes being the most commonly involved sites. Other affected areas include the uterine ligaments, rectovaginal septum, cul-de-sac, and pelvic peritoneum. Additionally, extra-pelvic endometriosis has been reported in various organs such as the gastrointestinal tract, bladder, kidneys, omentum, lymph nodes, lungs, pleura, extremities, umbilicus, hernia sacs, abdominal wall, heart, and even the brain[5-8]. The gastrointestinal tract is the most frequent site of extra-pelvic endometriosis, with intestinal involvement occurring in ~3–37% of women diagnosed with endometriosis, particularly affecting the rectum and sigmoid colon. However, <10% of intestinal cases occurs in the small intestine[8-11]. The manifestations of intestinal endometriosis are typically associated with the size and position of the lesion presenting with various symptoms, such as nausea, vomiting, abdominal pain, abdominal distension, tenesmus, painful defecation, rectal bleeding, and rarely intestinal obstruction. However, it could be asymptomatic[4,12]. Endometriosis of the distal ileum is a rare cause of intestinal obstruction, occurring in 7–23% of cases of intestinal endometriosis[13]. The diagnosis of DIE still represents a challenge and is usually confirmed during surgery or after complications like bowel obstruction, perforation, or ileocecal intussusception[1]. Surgical resection is the optimal treatment of small bowel obstruction (SBO) secondary to endometriosis; this intervention serves as both a diagnostic and a therapeutic procedure[4]. The choice between lesion resection (linear nodulectomy) and segmental resection depends on the size and extent of the endometriotic involvement. Segmental resection has been the most commonly performed surgical approach, particularly in cases with significant bowel involvement[14]. It is generally indicated for larger or more invasive lesions, particularly those exceeding 2.25 cm in length or involving >27% of the bowel circumference[15]. Conversely, lesion resection may be appropriate for smaller and less infiltrative nodules Herein, we discuss a patient’s case who came with abdominal pain and signs of SBO. Ultimately, she ended up with the diagnosis of DIE. Therefore, this report aims to emphasize the importance of having a suspicion of DIE as a potential cause of chronic and/or acute abdominal pain, which may initially be overlooked. Informed consent was obtained from the patient for publication of this case report and any accompanying images. This work has been reported in line with the SCARE criteria[16]. Case presentation A 43-year-old Syrian female presented to the emergency department with generalized abdominal pain lasting 24 h, accompanied by vomiting, fever, and bloating. Her medical history was significant for infertility and chronic abdominal pain, constipation, and cramps, which had previously been attributed to irritable bowel syndrome. She denied any prior medication use, surgeries, or known allergies. Clinical examination revealed significant abdominal distention and marked tenderness, most notably in the right iliac fossa. Laboratory tests, including a complete blood cell count (CBC), indicated leukocytosis with a white blood cell count of 17 × 103/μl (normal range: 4.4–11 × 103/μl) with elevated neutrophils (90%) (normal range: 40–70%). Additionally, there was an increased C-reactive protein (CRP) level of 120 mg/l (normal range: up to 5.0 mg/l). Urinalysis revealed a mild urinary infection, indicating 20–30 white blood cells/ml (normal range: 0–1 cells/ml), which did not explain the patient’s severe abdominal symptoms. Abdominal ultrasound imaging revealed a moderate amount of free fluid and dilatation of the intestinal loops. Abdominal X-rays (AXR) in the standing position showed the presence of air-fluid levels and dilated small bowel loops (Fig. 1). Figure 1.: Abdominal X-rays in the standing position showing the presence of air-fluid levels and dilated small bowel loops. A surgical laparotomy was performed and revealed a moderate amount of free fluid and dilatation within the small intestine. At ~10 cm from the ileocecal junction, an edematous segment of the terminal ileum was found to be volvulated, causing obstruction with proximal dilatation (Fig. 2). Figure 2.: Gross view of the small intestine showing a volvulated edematous segment of the terminal ileum. The affected intestinal segment was resected, followed by establishing an end-to-end ileo-ileal anastomosis to restore the continuity of the small intestine. The anastomosis was performed using a hand-sewn, two-layer technique, with the inner layer sutured continuously and the outer layer closed with interrupted sutures. 3-0 Vicryl was used as the suture material. This was done through a McBurney incision. Microscopic examination of the resected segment revealed benign small intestinal mucosa and wall. Variable amounts of inflammatory cells were found, along with cystic endometrial glands surrounded by endometrial stroma (Fig. 3). Based on these findings, the diagnosis of ileal endometriosis was confirmed. The patient achieved a full recovery after the surgery and experienced a significant improvement in quality of life on follow-up, with resolution of her chronic abdominal symptoms. Afterward, she was counseled to visit a gynecologist as the endometriosis could be causing her infertility. Figure 3.: Microscopic examination of the resected segment reveals cystic endometrial glands surrounded by endometrial stroma.

and conclusions Up to 15% of women who are menstruating typically have endometriosis. Between 3% and 37% of women have endometriosis-related gastrointestinal symptoms in the rectum, terminal ileum, and sigmoid colon[13]. Relative pelvic distribution of endometriosis indicates that the rectosigmoid colon, small bowel, appendix, and cecum are affected in 50–90%, 2–16%, 3–18%, and 2–15% of patients, respectively[1]. Depending on where the disease is involved, intestinal endometriosis symptoms might include nausea, vomiting, stomach discomfort, abdominal distension, tenesmus, painful defecation, and rectal hemorrhage. These symptoms may resemble those of other intestinal disorders, including irritable bowel syndrome, inflammatory bowel disease, and bowel tumors[17]. The present case illustrates how chronic symptoms—previously attributed to irritable bowel syndrome—can mask an underlying intestinal endometriosis that eventually presents with an acute surgical emergency. Several cases of distal ileal endometriosis leading to small bowel obstruction have been reported in the literature. Table 1 provides a summary of six cases of endometriosis-related SBO involving the distal ileum, showing the variability in clinical presentation, diagnostic approaches, and management. Table 1 - Reported cases of endometriosis-related small bowel obstruction involving the distal ileum. Diffuse, abdominal pain most prominent in the left lower quadrant, nausea and vomit Terminal ileum, ovaries sigmoid colon, appendix CT scan, exploratory laparotomy Resection of involved segment with primary anastomosis The underlying pathophysiology of SBO can be attributed to the the fibrosis of endometriosis in the muscularis and serosa of the small bowel that can result in stricture, which may lead eventually to obstruction and perforation[24]. A thickened, stenosing mass can be identified with the use of a CT scan; however, endometrial deposits are seldom seen by this method. When it comes to endometriosis diagnosis, magnetic resonance imaging (MRI) offers high sensitivity (77–93%). However, when examining potential endometrial deposits on the intestinal wall, diagnostic laparoscopy and biopsy are still considered the gold standard of care. It permits excision if necessary and views of the pelvic and abdominal organs[4]. Endometriosis can be treated with both surgical and pharmaceutical modalities. The majority of cases with symptomatic gastrointestinal endometriosis are surgically treated due to the challenges in establishing a preoperative diagnosis[25,26]. A recent meta-analysis indicated no difference in the short-term clinical outcomes of single-incision versus traditional laparoscopic surgery in terms of mortality, morbidity, operative time, blood loss, the rate of conversion to open surgery, perioperative complications, and hospital stay duration[25]. When the preoperative diagnosis is uncertain, the surgical approach allows for both accurate diagnostics and treatment at the same time. On the other hand, the pharmacological management of confirmed cases of bowel endometriosis without obstructive symptoms has been proven to be effective. A select variety of pharmacological agents, such as aromatase inhibitors[27], gonadotropin-releasing hormone agonists, and nonsteroidal anti-inflammatory drugs (NSAIDs), in conjunction with oral contraceptives, have been effective in reducing the severity and symptoms of endometriosis by inducing a hypoestrogenic state and endometrial atrophy[4]. In this case, due to the severe acute abdominal pain on presentation that was accompanied by signs of small bowel obstruction, an emergent surgical approach was preferred. This study has several limitations. First, it is a single case study, which may not provide findings that are generalizable to all patients with gastrointestinal endometriosis. Additionally, the diagnosis was confirmed only during surgery, emphasizing the challenges in making a preoperative confirmed diagnosis. Furthermore, the symptoms of intestinal endometriosis can vary widely among individuals, and the specific presentation in this case may not reflect the complete spectrum of the condition. In conclusion, even though it is rare, it is important to consider gastrointestinal endometriosis in the differential diagnosis of chronic or acute abdominal pain and small bowel obstruction, especially in women of reproductive age with a history of infertility or endometriosis. The unique aspect of this case lies in the location of the endometriosis in the terminal ileum, which is an uncommon site for endometriosis-related small bowel obstruction. Ethical approval Patient anonymity is maintained throughout this manuscript. Ethical approval and consent were obtained for publication from the patient. Consent Written informed consent was obtained from the patient for publication and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request. Sources of funding There was no funding for this paper. Author contributions M.A. participated in writing the original manuscript, prepared figures, and revised the final manuscript. A.A. participated in writing the original manuscript. H.H. participated in writing the original manuscript and collecting data. A.Q participated in writing the original manuscript. B.A. participated in writing the original manuscript. S.Y. was involved in establishing the diagnosis and patient care and revised the manuscript. S.M.B. was involved in establishing the diagnosis and patient care and revised the final manuscript. All authors read and approved the final manuscript. Conflicts of interest disclosure None declared. Research registration unique identifying number Not applicable. Guarantor Sari Medhat Barshiny

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