{"paper_id":"4f7df779-4f03-4e21-90e6-d3ba64c3d070","body_text":"Intussusception of the Cecal Appendix\nSecondary to Endometriosis: Case Report\nSofiaM a r a s c aG i o n g o1 Gabriela Pinho Fillmann 1 Marcelo Garcia Toneto 1\nLúcio Sarubbi Fillmann 1 Alessandro Batista Soares 1 Mariana Tanus Stefani 1 Marina Tonin 1\nAna Laura Avila Caumo 1 Irina Maria Ayala Lopez 1\n1 Ponti fical Catholic University of RS, Porto Alegre, RS, Brazil\nJ Coloproctol 2025;45(1):s00451802596.\nAddress for correspondence Gabriela Pinho Fillmann, Ponti fical\nCatholic University of RS, Porto Alegre, RS, Brazil\n(e-mail: G. fillmann@edu.pucrs.br).\nIntroduction\nThe intussusception of the appendix is the invagination of\nthe appendix into the cecum, accompanied by changes in\nperistalsis.\n1 This condition is considered a rare finding, with\nan incidence of 0.01%. In the clinical assessment, the main\nsymptoms presented by the patients are abdominal pain,\nvomiting, and blood in the rectum. However, their presenta-\ntion varies. Thus, while some people have acute symptoms\nsimilar to those of appendicitis, other patients have\nKeywords\n► appendix\n► intussusception\n► adults\n► endometriosis\nAbstract Introduction Appendicular intussusception is the invagination of the appendix into\nthe cecum. Endometriosis is the presence of endometrial tissue outside the uterine\ncavity, and it has a variable clinical pict ure, presenting as acute appendicitis or\nintestinal obstruction due to intussusception. The aim of the present paper is to\nreport a case of cecal appendix intussusception secondary to endometriosis, since this\nis an extremely rare condition and there are few studies on the subject in the literature.\nCase Report We herein report the case of a 37-year-old female patient with deep\nendometriosis and abdominal pain in the righ t iliac fossa associated with menstruation.\nThere was an intraluminal formation in the cecum, in the appendicular ostium.\nMagnetic resonance imaging showed foci compatible with ectopic endometrium in\nthe uterus, rectum, and sigmoid colon. The cecal appendix was enlarged. Resection of\nthe terminal ileum and cecum was performed, and the surgical specimen led to the\ndiagnosis of appendicular intussusception and endometriosis of the cecal appendix.\nDiscussion In adults, the most common etiologies of appendicular intussusception\nare endometriosis, mucocele, carcinoid tumors, and adenocarcinomas. In patients with\nendometriosis, involvement of the cecal appendix is infrequent. The de finitive diagno-\nsis is established by analyzing the surgical specimen, and imaging tests play a limited\nrole in ruling out malignancies. Laparoscopic cecectomy is usually chosen, preserving\nthe ileocecal valve. Hormone suppression therapy can prevent the recurrence of\nintestinal endometriosis.\nConclusion Appendicular intussusception, although an infrequent pathology, is an\noccurrence that requires surgical treatment and, in patients with endometriosis, it is\nimportant to include this hypothesis in the differential diagnosis.\nreceived\nAugust 13, 2024\naccepted after revision\nNovember 25, 2024\nDOI https://doi.org/\n10.1055/s-0045-1802596.\nISSN 2237-9363.\n© 2025. The Author(s).\nThis is an open access article published by Thieme under the terms of the\nCreative Commons Attribution 4.0 International License, permitting copying\nand reproduction so long as the original work is given appropriate credit\n(https://creativecommons.org/licenses/by/4.0/).\nThieme Revinter Publicações Ltda., Rua Rego Freitas, 175, loja 1,\nRepública, São Paulo, SP, CEP 01220-010, Brazil\nTHIEME\nCase Report 1\nArticle published online: 2025-03-20\n\nintermittent symptoms, which can persist for weeks or\nmonths. The most common cause of appendix intussuscep-\ntion in adult patients is endometriosis, which accounts for\napproximately 33% of the cases.\n2\nEndometriosis can be de fined as the presence of endome-\ntrial glands and stroma outside the uterine cavity. This\ncondition affects around 10% of women who menstruate,\nand it is present in approximately 70% of the cases of chronic\npelvic pain and in 50% of the cases of infertility.\n3 The clinical\nassessment of endometriosis is variable, and many individu-\nals are asymptomatic. Most of the symptomatic cases tend to\npresent moderate to severe pelvic pain. Endometriosis in the\nappendix can result in an asymptomatic condition, but it can\nalso present as acute appendicitis, intestinal obstruction due\nto intussusception, lower gastrointestinal bleeding, or intes-\ntinal perforation.\n3\nGiven that intussusception of the appendix due to endo-\nmetriosis is an extremely rare finding, there is a scarcity of\nstudies on the subject in the literature, and there is a need for\nmore publications on the subject.2 We herein report a case of a\npatient with intussusception of the cecal appendix secondary\nto endometriosis.\nCase Report\nA 37-year-old, White, single, female patient, diagnosed with\ndeep endometriosis 3 years before, consulted with a colo-\nproctologist complaining of recurrent abdominal pain in the\nright iliac fossa associated with her menstrual period for\n2 years, with no change in her bowel habits, bleeding or\nconstitutional symptoms. The physical examination revealed\npain on palpation of the right iliac fossa, with no signs of\nperitonitis. The patient had undergone a colonoscopy two\nyears before, when the symptoms began, in which a tubular\nadenomatous polyp with low-grade dysplasia was identi fied\nin the sigmoid, with no other endoscopic findings.\nThe patient returned for a review, presenting partial relief\nof the symptoms after starting the treatment for endometri-\nosis with hormone blockade, but continued to present\ndiscomfort in her right iliac fossa, with the same character-\nistics as those at the start of the investigation. A new\ncolonoscopy was recommended to assess deep endometri-\nosis and as a follow-up after resection of the adenomatous\npolyp. Upon further examination, the endoscope was intro-\nduced up to the terminal ileum, showing an intraluminal\nformation 4 cm long in the cecum, near the concentration of\nthe tapeworms, apparently in the region of the appendicular\nostium, with a slightly enanthematous mucosa, but with the\narchitecture of the crypts preserved on biopsy (\n►Fig. 1 ). A 5-\nmm sessile polyp was also identi fied in the sigmoid colon,\nwhich was removed. The material collected was sent for an\nanatomopathological examination. The sessile sigmoid polyp\nturned out to be a tubular adenoma with low-grade dyspla-\nsia, and the biopsy of the cecal lesion identi fied tissue\ncorresponding to the cecal appendix.\nThe next step in the investigation involved a magnetic\nresonance imaging (MRI) scan to evaluate the colonoscopic\nfindings, which showed hyperintense foci, compatible with\nectopic endometrium in the uterine junctional zone, with\nother foci involving part of the uterus and ligaments, the\nanterior wall of the rectum, and the sigmoid colon, with\nparietal thickening and vegetation, possibly related to colon-\nic endometriosis (\n►Fig. 2 ). The cecal appendix had diffusely\nthickened walls and increased volume (2.0 cm in diameter),\nwhich may correspond to a case of mucocele, and its base\nprojected into the lumen of the cecum.\nThe main diagnostic hypothesis was of a pathological\nappendix with suspected intussusception, and the patient\nwas referred for surgery. A right laparoscopic ileocolectomy\nwas performed, with resection of 15 cm of the terminal ileum\nand cecum (\n►Fig. 3 ) and primary ileocolic anastomosis. The\nanatomopathological results of the surgical specimen led to\nthe diagnosis of intussusception of the cecal appendix with a\nmarked chronic in flammatory process and associated endo-\nmetriosis of said appendix. The resection margins were\nviable, with four mesocolic and mesenteric lymph nodes\nshowing reactive lymphoid hyperplasia and sinusoidal con-\ngestion. The patient progressed satisfactorily and was dis-\ncharged from the hospital on the fifth postoperative day,\nwithout any complications. At the 60-day review, she\nshowed a signi ficant improvement in her right iliac fossa\npain.\nDiscussion\nIntussusception of the cecal appendix is a rare, dif ficult-to-\ndiagnose condition with an incidence of 0.01%. 1,2 It is more\nprevalent in the adult population (76%), with a predomi-\nnance in females (70%), and it is more commonly associated\nwith cases of endometriosis, appendicular mucocele, Crohn ’s\ndisease, adenoma, and adenocarcinoma. 2,4\nThe clinical picture of appendicular intussusception is\nvariable, ranging from asymptomatic to nonspeci fic acute\nand chronic symptoms, or those that can mimic acute\nappendicitis.5 Chaar et al. 2 have reported that nonspeci fic\nchronic symptoms are more common, occurring in 63% of the\nFig. 1 Lesion identi fied in the cecum during colonoscopy: 4-cm long\nintraluminal formation, apparently in the region of the appendicular\nostium, with slightly enanthematous mucosa.\nJ Coloproctol Vol. 45 No. 1/2025 © 2025. The Author(s).\nCecal Appendix Intussusception Secondary to Endometriosis Giongo et al.2\n\n\ncases, with abdominal pain (78%), vomiting and melena\n(26%), and hematochezia (28%) standing out.\nAccording to Costa et al., 5 the etiology of appendicular\nintussusception may be related to anatomical factors, such\nas a large proximal appendicular lumen, hyperperistalsis, a\nfully mobile appendix or a thin mesoappendix. Among the\nfactors that trigger intussusception are foreign bodies, feca-\nliths, polyps, adenocarcinomas, carcinoid tumors, parasites,\nendometriosis, and lymphoid hyperplasia.\n1,2,5 In adults, en-\ndometriosis is the most common etiology, responsible for 33%\nof the cases, while approximately 7% of the cases are caused by\ncarcinoid tumors, and 6%, by adenocarcinomas.\n2\nIt is estimated that endometriosis affects around 10% of\nwomen of reproductive age, causing involvement of the\nintestinal tract in 3.8 to 37% of the patients. 1,6 Most of these\ncases develop the disease in the rectum and rectosigmoid\njunction, with involvement of the cecal appendix being rare,\noccurring in approximately 3% of cases.\n1,7 Endometrial tissue\nin the intestine most often affects the serous and muscular\nlayers, causing adhesions and hyperplasia of the smooth\nmuscle; the mass formed is propelled by peristalsis and, if\nit affects the cecal appendix, it can cause its intussusception\nin the cecum, as in the case herein presented.\n2,8\nThe suspicion of endometriosis as the etiology of intesti-\nnal intussusception arises mainly due to a previous diagnosis\nof the disease or a clinical history of complaints such as\ndysmenorrhea, dyspareunia, chronic pelvic pain, and dyski-\nnesia.\n6 Based on this hypothesis, a targeted investigation into\ndifferential diagnoses is essential.\nThe diagnosis of cecal appendix intussusception is an\narduous task, and the condition is often confused with neo-\nplasms.2,5 In addition, most cases are only identi fied during\nsurgery (57%), while preoperative diagnosis (32%) is less\nfrequent.\n2 Chaar et al. 2 have reported that preoperative\ndiagnosis after the 2000s with more improved imaging tests\nhas resulted in increased sensitivity.\nInitially, ultrasound (US), computed tomography (CT) of\nthe abdomen, radiography with barium enema and endo-\nscopic studies can be performed.\n5,9 During colonoscopy, the\nlesion is usually observed as a polypoid mass covered by\nnormal mucosa and a central depression in the ori fice.5\nComputed tomography is more accurate than US, although\nboth tests help in the diagnosis. 3 Magnetic resonance imag-\ning has been used 10 for better preoperative assessment and\ndifferentiation from mucoceles, showing hyperintense foci\ncompatible with ectopic endometrium, suggestive of colonic\nendometriosis.\nHowever, imaging tests and endoscopic biopsies play a\nlimited role in ruling out malignancies, and the suspicion of\nendometriosis is not well assessed in colonoscopy biopsies,\nsince most endometrial implants are located in the outer\nlayers of the intestinal wall.\n8 In these cases, endoscopic\nbiopsy often shows normal mucosa, as in the case herein\ndescribed. Therefore, the de finitive diagnosis, in most cases,\nis established by surgical approach and complete analysis of\nthe specimen.\n2\nIn general, laparoscopic cecectomy is chosen, with pres-\nervation of the ileocecal valve, but it may also be necessary to\nperform ileocecectomy or right hemicolectomy if there is an\nFig. 3 Surgical specimen with resection of the terminal ileum and\ncecum and intussusception of a pathological appendix. The segments\nof the small and large intestines measure 6.5 cm and 4.5 cm in length\nrespectively and have a grayish-violaceous serosa with congested\nvessels, in which you can see an area of mesoretraction. At the\nopening, close to the ileocecal valve, an area of protrusion lesion can\nbe seen in the lumen of the cecum, in which the cecal appendix can be\nidenti fied, measuring approximately 4.5 cm in length and 1.5 cm in\nmaximum diameter, with a shiny gray serosa and mucosa with\npreserved folding.\nFig. 2 Total abdomen magnetic resonance imaging scan in a T2-weighted fast-spin echo (T2 FSE) sequence. ( A) Coronal section with hyposignal\nin the cecal appendix (blue arrow). ( B) Axial section with hyposignal in the cecal appendix (green arrows).\nJ Coloproctol Vol. 45 No. 1/2025 © 2025. The Author(s).\nCecal Appendix Intussusception Secondary to Endometriosis Giongo et al. 3\n\n\nassociation with ileocecal or ileoileal intussusception. Lapa-\nroscopy also enables the assessment of the peritoneal cavity\nin search of other foci of endometriosis.\n9\nIn addition to surgery, the treatment after the de finitive\ndiagnosis of endometriosis etiology may involve hormone\nsuppression therapy, which presents bene fits mainly in\npreventing recurrences of intestinal endometriosis. In these\ncases, the first line of treatment is low dose progesterones or\ncombined oral contraceptives, both of which present few\nside effects and are inexpensive. Follow-up with a gynecolo-\ngist and coloproctologist is essential.\n6\nConclusion\nIntussusception is a rare occurrence that is dif ficult to\ndiagnose in the adult population, and it involves the colon\nless often than the small intestine. Despite the rarity of the\nclinical picture, the case herein reported, of a female patient\nwith cecal appendix intussusception due to endometriosis,\nwho underwent surgical treatment with good postoperative\nevolution, demonstrates the need to include this hypothesis\nin the differential diagnosis of patients with similar symp-\ntoms of recurrent abdominal pain in the right iliac fossa\nrelated to the menstrual period.\nAuthors’ Contributions\nSofia Marasca Giongo was responsible for analyzing and\ninterpreting all data, as well as contributing to the design\nof the study. Gabriela Pinho Fillmann was involved in the\nanalysis and interpretation of data, as well as in writing\nthe manuscript. Marcelo Garcia Toneto and Lúcio Sarubbi\nFillmann supervised the manuscript and conducted the\nfinal review. Alessandro Batista Soares, Mariana Tanus\nStefani, Marina Tonin and Ana Laura Avila Caumo contrib-\nuted by analyzing and interpreting the data and assisting\nin writing the manuscript. Irina Maria Ayala Lopez ana-\nlyzed the images used in the article.\nFunding\nThe author(s) received no financial support for the\nresearch.\nConflict of Interests\nThe authors have no con flict of interests to declare.\nReferences\n1 L o p e zM P J ,C h a nV ,M e l e n d r e sM F ,L u t a n c oR .A p p e n d i c e a li n t u s s u s -\nception from endometriosis. BMJ Case Rep 2021;14(06):e241592\n2 Chaar CI, Wexelman B, Zuckerman K, Longo W. Intussusception of\nthe appendix: comprehensive review of the literature. Am J Surg\n2009;198(01):122–128\n3 Yoon J, Lee YS, Chang HS, Park CS. Endometriosis of the appendix.\nAnn Surg Treat Res 2014;87(03):144 –147\n4 Trefois C, Coche E. Appendiceal Intussusception Secondary to\nEndometriosis: A Rare Etiology of Right Lower Quadrant Abdom-\ninal Pain. J Belg Soc Radiol 2022;106(01):34\n5 Costa M, Bento A, Batista H, Oliveira F. Endometriosis-induced\nintussusception of the caecal appendix. 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