A Rare Cause of Bowel Obstruction Mimicking Colon Cancer: Endometriosis

Endometriosis; pelvic pain; ileus; rectal neoplasms; colectomy DOI: 10.5336/jcog.2021-85436 Correspondence: Sami AÇAR Department of General Surgery, University of Health Sciences Zeynep Kamil Maternity and Children Training and Research Hospital, İstanbul, Türkiye E-mail: [email protected] Peer review under responsibility of Journal of Clinical Obstetrics & Gynecology. Re ce i ved: 09 Jul 2021 Received in revised form: 14 Nov 2021 Ac cep ted: 01 Feb 2022 Available online: 08 Feb 2022 2619-9467 / Copyright © 2022 by Türkiye Klinikleri. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/). Turkiye Klinikleri Journal of Internal Medicine Journal of Clinical Obstetrics & Gynecology CASE REPORT 28 CASE REPORT A 41-year-old gravida 0 patient presented with pain in the form of pressure in the pelvic area and difficulty in stooling. Three different colonoscopic examinations were performed at 3 different gas- troenterology departments. In all these colonoscopic examinations, a mass lesion starting approximately 5 cm from the anal entrance was seen, and it extended along the rectal segment about 3-4 cm. According to the Paris classification, it was graded as Is+IIb. Due to the presence of a Kudo pit pattern V appearance in one area, it was not suitable for endoscopic resection ( Figure 1). All pathological evaluations were com- patible with the granulation tissue. In the pelvic area, a mass lesion 5x6 cm in diameter, with lobulated con- toured internal small air loculations, located on the expanded floor of the Douglas pouch was observed. Diffuse thickening up to 15 mm in a circular fashion at the rectal wall and the presence of many lymph nodes reaching 12 mm in diameter in the mesorectal plane were suspicious for cancer ( Figure 2). A surgi- cal treatment decision was made with oncological principles for a mass lesion located in the middle rec- tum with occlusive nature. Due to previous abdomi- nal surgery, coloanal anastamosis following laparotomic mesocolic and mesorectal rectosig- moidal resection, and loop ileostomy was performed ( Figure 3). In the definitive pathological evaluation, it was reported that the resection performed was close to complete, and reactive changes was determined in 18 lymph nodes which were surgically removed, and a mass lesion with a diameter of 5.5x5x4.5 cm was re- ported as endometriosis ( Figure 4). Ectopic polypoid endometrial tissue in the rectum was located in the en- tire muscular, submucosal and mucosal layers, and a large polypoid mass could be seen protruding to the lu- mina and subsequently occluding it. The exulcerated surface of the polyp with wide granulation tissue was shown microscopically ( Figure 5). Sami AÇAR et al. JCOG. 2022;32(1):27-31 28 FIGURE 1: Rectosigmoidoscopic view, a mass lesion with the features of being firm and smooth surfaced, including hyperemic areas that alm ost completely obstructing the rectal wall. FIGURE 2: (A) Transverse and (B) sagittal plane of the pelvic magnetic resonance images, respectively. A mass lesion 5x6 cm in diameter with lobulated counter internal small air loculations on the expanded floor of the Douglas pouch. 292929 Immunohistochemistry technique for CD10, PAX8, es- trogen alpha (ERα) and progesterone (PR) receptor, was performed to confirm the presence of endometrial stroma and glandular tissue ( Figure 6). An informed consent was obtained from the pa- tient for this case report.

The presence of intestinal endometriosis can be de- tected as a result of examinations performed for pelvic pain, presence of an unknown mass, and rec- tal bleeding. Mechanical bowel obstruction develops due to areas of endometriosis extending into the lumen in the rectosigmoid region and sigmoid colon. 7 Similarly, endometriosis foci can cause plication and angulation of the intestinal segment around it. 8 Intes- tinal obstruction with nodules extending from the Douglas pouch to the middle rectum is extremely rare. This is because the ampulla recti is large, cali- brated and flexible, and also due to its anterior face being covered by the peritoneum. Ono et al. have re- ported that the development of obstruction in the rec- tal wall occur because of narrowing of the lumen, ultimately due to the location of the endometriosis. Since the diagnosis of malignancy could not be con- firmed, proctectomy was performed. 9 Lenz et al. also presented a case of endometriosis causing perfora- tion. 10 Sami AÇAR et al. JCOG. 2022;32(1):27-31 29 FIGURE 3: (A) Complete mesocolic and mesorectal excision specimen, (B) intra-abdominal view after resection. FIGURE 4: (A) The image shows, a mass completely obstructing the rectal lumen, (B) the view of the mass lesion right after surgically openning of the posterior rectal wall. The mass is located on the ventral side of the rectum. FIGURE 5: Endometriosis; replacing mucosa, submucosa and muscularis propria and protruding lumina of the rectum as a bulky mass (H&E, x20). Rectum-located endometriosis and rectal cancer cannot be differentiated frequently and therefore wide resections are performed. 11 The incidence of malig- nancy associated with endometriosis is 1%, and this probability is higher in foci with extraovarian loca- tion. Surgical treatment methods are erasion from in- testinal surface, discoid excision, and segmental bowel resection. The method of choice depends on the location of intestinal involvement, the number of foci on the intestinal surface, the depth of the infil- tration, and whether there is a stenosis in the gut lumen. 7 Avoiding segmental bowel resection, espe- cially in the presence of lesions close to the anal verge level, is important in terms of preventing pos- sible complications. Even in bowel resection, the presence of microscopic endometriosis foci at the level of 15% at the resection limits requires a careful evaluation at the decision-making stage. Roman et al. have reported that the long-term results of radical sur- gery are not prognostically brighter than conservative treatment, and the complication rates seem to be higher. 12 Vlek et al. have defined the transanal mini- mally invasive surgical technique for rectal en- dometriosis with deep localization. 13 Kazama et al. have stated that the rectal en- dometriosis area was prone to bleeding on endo- scopic examination. They have defined the presence of pits with flat microvessels in magnified narrow band imaging, and avascular areas at the tip of the papillary protrusions. 14 As eutopic endometrium, ec- topic glands express ER, PR, PAX-8 and stroma ex- press CD10. On the other hand, colonic glands are well known to express CDX2 which was regarded a highly sensitive marker of intestinal epithelium whereas PAX-8 was reactive for organs derived from mullerian duct. In our case, although H+E stained slides were pretty clear about endometriotic nature of the polypoid mass with stroma and glands, we con- firmed the diagnosis with the panel of above-men- tioned immunohistochemical stains and CDX2 was negative whereas others were all positive. Zondervan et al. have summarized the most im- portant reasons for the delay in diagnosis of en- dometriosis. These are the absence of specific complaints and biological markers, insufficient aware- ness and occasional normalization of the findings. 15 The uncertainty in diagnosis cannot clarify which sur- gical intervention should be at the decision-making stage. Because, in the presence of possible rectal can- cer, mesorectal excision and neoadjuvant treatment plan comes to the fore. Failure to diagnose prevents further examination. Insufficient surgical treatment in the presence of cancer has a negative effect on possi- ble local recurrence and survival. On the other hand, wide resection in the presence of a benign condition increases the complication rate. In case of stoma open- ing, quality of life deteriorates, and secondary surgi- cal interventions are required for closure. Sami AÇAR et al. JCOG. 2022;32(1):27-31 30 FIGURE 6: Positive (A) CD10, (B) PAX9, (C) ER, (D) PR immunohistochemical staining of endometriosis in rectum (IHC,x20). Despite the colonoscopic biopsies that were per- formed in different gastroenterology departments for 3 times; the endometriosis could not have been diag- nosed and the patient underwent a surgical interven- tion. It should be kept in mind that endometriosis may be the cause of ileus in women of childbearing age and should be considered in the differential diagnosis of both malignancy and diverticulosis coli. Source of Finance During this study, no financial or spiritual support was received neither from any pharmaceutical company that has a direct con- nection with the research subject, nor from a company that pro- vides or produces medical instruments and materials which may negatively affect the evaluation process of this study. Conflict of Interest No conflicts of interest between the authors and / or family mem- bers of the scientific and medical committee members or mem- bers of the potential conflicts of interest, counseling, expertise, working conditions, share holding and similar situations in any firm. Authorship Contributions Idea/Concept: Sami Açar; Design: Sami Açar; Control/Supervi- sion: Murat Api, Handan Çetiner; Data Collection and/or Pro- cessing: Sami Açar, Handan Çetiner, Erman Çiftçi; Analysis and/or Interpretation: Sami Açar, Murat Api; Literature Review: Sami Açar, Erman Çiftçi; Writing the Article: Sami Açar; Criti- cal Review: Murat Api; References and Fundings: Sami Açar;

Sami Açar. Sami AÇAR et al. JCOG. 2022;32(1):27-31 31 1. Shafrir AL, Farland LV, Shah DK, Harris HR, Kvaskoff M, Zondervan K, et al. Risk for and consequences of endometriosis: a critical epidemio- logic review. Best Pract Res Clin Obstet Gynaecol. 2018;51:1-15. [Cross- ref] [PubMed] 2. Vercellini P, Viganò P, Somigliana E, Fedele L. Endometriosis: patho- genesis and treatment. Nat Rev Endocrinol. 2014;10(5):261-75. [Cross- ref] [PubMed] 3. De Cicco C, Corona R, Schonman R, Mailova K, Ussia A, Koninckx Pr. Bowel resection for deep endometriosis: a systematic review. BJOG. 2011;118(3):285-91. [Crossref] [PubMed] 4. Meuleman C, Tomassetti C, D'Hoore A, van Cleynenbreugel BV, Pen- ninckx F, Vergote I, et al. Surgical treatment of deeply infiltrating en- dometriosis with colorectal involvement. Hum Reprod Update. 2011;17(3):311-26. [Crossref] [PubMed] 5. Johnson NP, Hummelshoj L, Adamson GD, Keckstein J, Taylor HS, Abrao MS, et al. World Endometriosis Society consensus on the classification of endometriosis. Hum Reprod. 2017;32(2):315-24. [Crossref] [PubMed] 6. Becker CM, Gattrell WT, Gude K, Singh SS. Reevaluating response and failure of medical treatment of endometriosis: a systematic review. Fer- til Steril. 2017;108(1):125-36. [Crossref] [PubMed] [PMC] 7. Nezhat C, Li A, Falik R, Copeland D, Razavi G, Shakib A, et al. Bowel endometriosis: diagnosis and management. Am J Obstet Gynecol. 2018;218(6):549-62. [Crossref] [PubMed] 8. Vercellini P, Sergenti G, Buggio L, Frattaruolo MP, Dridi D, Berlanda N. Advances in the medical management of bowel endometriosis. Best Pract Res Clin Obstet Gynaecol. 2021;71:78-99. [Crossref] [PubMed] 9. Ono H, Honda S, Danjo Y, Nakamura K, Okabe M, Kimura T, et al. Rec- tal obstruction due to endometriosis: a case report and review of the Japanese literature. Int J Surg Case Rep. 2014;5(11):845-8. [Crossref] [PubMed] [PMC] 10. Lenz J, Fiala L, Chvatal R, Tibon J, Uncapher L, Kavka M, et al. Rectal perforation caused by deep infiltrating endometriosis in non-pregnant woman: case report and short review of the literature. Ann Ital Chir. 2019;8:S2239253X19029360. [PubMed] 11. Rana R, Sharma S, Narula H, Madhok M. A case of recto-sigmoid en- dometriosis mimicking carcinoma. Springerplus. 2016;5:643. [Crossref] [PubMed] [PMC] 12. Roman H, Milles M, Vassilieff M, Resch B, Tuech JJ, Huet E, et al. Long- term functional outcomes following colorectal resection versus shaving for rectal endometriosis. Am J Obstet Gynecol. 2016;215(6):762.e1-9. [Crossref] [PubMed] 13. Vlek SL, Lier MCI, Koedam TWA, Melgers I, Dekker JJML, Bonjer JH, et al. Transanal minimally invasive rectal resection for deep endometrio- sis: a promising technique. Colorectal Dis. 2017;19(6):576-81. [Crossref] [PubMed] 14. Kazama S, Hiramatsu T, Kuroda K, Hongo K, Watanabe Y, Tanaka T, et al. A case of unique endoscopic findings of intestinal endometriosis exposed to the mucosa: aggregation of papillary protruded bulges from the submucosal elevation of the rectum. Clin J Gastroenterol. 2019;12(2):166-70. [Crossref] [PubMed] 15. Zondervan KT, Becker CM, Missmer SA. Endometriosis. N Engl J Med. 2020;382(13):1244-56. [Crossref] [PubMed] REFERENCES