Strangulated appendiceal intussusception caused by isolated endometriosis

246 Copyright © 2021 The Korean Society of Emergency Medicine Strangulated appendiceal intussusception caused by isolated endometriosis Seokyoun Lee1, Junhee Lee2, Keunyoung Kim3 1 Department of Surgery, Wonkwang University Sanbon Hospital, Wonkwang University School of Medicine, Gunpo, Korea 2 Department of Emergency Medicine, Wonkwang University Sanbon Hospital, Wonkwang University School of Medicine, Gunpo, Korea 3 Department of Surgery, Wonkwang University Hospital, Wonkwang University School of Medicine, Iksan, Korea Clin Exp Emerg Med 2021;8(3):246-248 https://doi.org/10.15441/ceem.20.026 eISSN: 2383-4625 Image Received: 16 March 2020 Revised: 18 May 2020 Accepted: 16 June 2020 Correspondence to: Seokyoun Lee Department of Surgery, Wonkwang University Sanbon Hospital, Wonkwang University School of Medicine, 321 Sanbon-ro, Gunpo 15865, Korea E-mail: [email protected] ORCID https://orcid.org/0000-0001-7245-8449 Lee S, Lee J, Kim K. Strangulated appendiceal intussusception caused by isolated endometriosis. Clin Exp Emerg Med 2021;8(3):246-248. https://doi. org/10.15441/ceem.20.026 This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (https:// creativecommons.org/licenses/by-nc/4.0/). What is already known Appendiceal endometriosis can present strangulated intussusception. What is new in the current study Appendiceal intussusception secondary to endometriosis should be considered in the differential diagnosis when female patients of reproductive age present with clinical symptoms of acute appendicitis. A 33-year-old female was admitted to our emergency department for right lower quadrant ab- dominal pain, which was not associated with her menstrual cycle, and nausea, which had per- sisted for 3 days. She denied any prior history of abdominal pain, dysmenorrhea, menorrhagia, or dyspareunia. The patient had no family history of endometriosis. Abdominal computed tomogra- phy revealed appendiceal intussusception (AI) and invagination of the periappendiceal fat into the cecum (Fig. 1). She underwent laparoscopic partial cecectomy with the presumed diagnosis of AI (Fig. 2). The ovaries and fallopian tubes were normal, with no endometrial spot. The invert- ed appendix was completely necrotic (Fig. 3). Final surgical pathology was positive for gangre- nous intussusception of the appendix, with endometrial deposits (Fig. 4). Adult colonic intussusception is caused by either malignant tumors (64%–87% cases) or be- nign tumors (29%–33%). The most common causes are adenocarcinomas and lymphomas among malignant tumors and lipomas, leiomyomas, adenomatous polyps, and endometriosis among be- nign tumors.1 AI is extremely rare, with reported rates of 0.01% in patients who undergo appen- dectomy.2 AI secondary to endometriosis is also extremely rare, with less than 30 cases reported. However, those cases involved endometriosis on other organs and the appendix. Only three cases involving isolated appendiceal endometriosis are reported in the literatures.3 Patients with AI can range from being asymptomatic to presenting symptoms mimicking acute appendicitis.4 Preop- erative diagnosis is difficult owing to its rarity. Although endometriosis is reported to be the lead point of AI, there are limited descriptions on imaging findings. Computed tomography findings include an enlarged appendix with an inverted appendiceal tip or a tumor protruding into the 247Clin Exp Emerg Med 2021;8(3):246-248 Seokyoun Lee, et al. cecum.5 This case highlights that isolated endometriosis without involvement of reproductive organs can present as a strangulated AI. AI secondary to endometriosis should be considered in the dif- ferential diagnosis when female patients of childbearing age present with clinical symptoms of acute appendicitis and radio- logic findings of intussusception, even before obtaining a patho- logical result. CONFLICT OF INTEREST No potential conflict of interest relevant to this article was re- ported. ACKNOWLEDGMENTS This paper was supported by Wonkwang University in 2020. REFERENCES 1. Lee SY, Park WC, Lee JK, Kang DB, Kim Y, Yun KJ. Laparoscopic treatment of adult sigmoidorectal intussusception caused by a mucinous adenocarcinoma of the sigmoid colon: a case re- port. J Korean Soc Coloproctol 2011;27:44-9. 2. Collins DC. 71,000 Human appendix specimens. a final report, summarizing forty years’ study. Am J Proctol 1963;14:265-81. Fig. 1. The abdominal computed tomography. (A) Axial view and (B) coronal view. These images show a low-density tubular structure (5×1.6 cm in diameter) in the cecum near the appendiceal orifice, and focal in- vagination of the peri-appendiceal fat to the cecum (white arrow). A B Fig. 3. Surgical specimen. Appearance of the specimen resected by lap- aroscopic partial cecectomy. The inverted appendix (70×15 mm in size) is completely necrotic at the appendiceal orifice (inside the aspect of the cecum denoted by a white arrow). Fig. 4. High-power view of endometriosis within the appendix. Endome- trial glands and stroma within the muscular layer, around the appendicu- lar mucosa (hematoxylin and eosin, × 200). Fig. 2. Surgical findings. During laparoscopic exploration, the appendix was not macroscopically visible in the right iliac fossa, but it was locat- ed in the cecum (white arrow), which showed no possibility for reduc- tion (black arrow). Terminal ileum denoted by asterisk. 248 www.ceemjournal.org Strangulated appendiceal intussusception 3. Manoharan B, Haider AS, Samira AM, Bharathan B, Parra- Blanco A. Isolated appendiceal endometriosis resulting in in- tussusception. Oxf Med Case Reports 2018;10:349-52. 4. Salehzadeh A, Scala A, Simson JN. Appendiceal intussuscep- tion mistaken for a polyp at colonoscopy: case report and re- view of literature. Ann R Coll Surg Engl 2010;92:W46-8. 5. Hanly AM, Ryan EM, McNamara DA. Appendiceal intussuscep- tion and endometriosis mimicking colorectal cancer. Colorectal Dis 2011;13:225-6.