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Adult Intussusception Caused by Cecal Lymphangioma: A Case Report/Cekal Lenfanjioma Bagli Eriskin Invajinasyon: Olgu Sunumu.

Introduction

Lymphangioma is a benign congenital tumor in the lymphatic system arising from a rare developmental anomaly. It is usually seen in childhood, with 80-90% of cases detected in the first five years of life, and rarely occurs in adulthood. They are most commonly located in the head, neck, and axilla. In rare cases they may also be found in the abdomen. (1,2) Gastrointestinal involvement occurs in less than 1% of cases and can cause bowel invagination. Intussusception is rare in adults, accounting for approximately 1-3% of all bowel obstructions. (3) In this report, we present a rare case of ileocecal intussusception due to intestinal lymphangioma in an adult patient.

Case Report

A 23-year-old woman presented to our emergency department with complaints of abdominal pain and diarrhea starting one week earlier. Her medical and family histories were unremarkable. On physical examination, she exhibited right lower quadrant abdominal tenderness. Her blood pressure was 100/60 mmHg, pulse was 96/min, and body temperature was 36.7 [degrees]C. Laboratory tests were normal except for elevated C-reactive protein level (2.9 mg/dL). Air-fluid level was observed in the small intestine

on standing anteroposterior X-ray. Abdominal ultrasound and computed tomography showed that the terminal ileum was invaginated into the cecum (Figure 1). Emergency laparotomy was performed due to escalation of the patient's abdominal pain and development of acute abdomen. The ileocecal intussusception and a solid mass in the cecum were detected during the laparotomy. After manual reduction, we observed that the invaginated small intestine loop had diminished perfusion, and ileocecal resection was performed. On macroscopic examination of the resected material, a 4x4x2 cm polypoidal lesion was observed at the cecal base, ulcerating the mucosa but limited to the submucosa (Figure 2). Microscopic examination revealed an endothelium-lined cyst divided by fibrous septa under a normal mucosal membrane (Figure 3). Histopathological diagnosis was reported as cystic lymphangioma.

The patient was discharged on postoperative day 6. No recurrence was detected in colonoscopy or computed tomography in follow-up at postoperative 3 and 9 months.

Discussion

Lymphangiomas are benign congenital tumors of the lymphatic system and are divided into three classes: capillary, cystic, and cavernous. (4) Cystic lymphangiomas are the most common. Cystic lymphangiomas have large endothelium-lined cystic spaces, often with thick walls, and collagen and smooth muscle are present at various proportions. Patients with lymphangioma are usually asymptomatic. Symptomatic patients may experience abdominal pain, vomiting, diarrhea, constipation, obstruction, invagination, lower gastrointestinal hemorrhage, and protein-losing enteropathy. (5) The diagnosis of intraabdominal lymphangioma can be facilitated by barium enema, colonoscopy, endoscopic ultrasonography, abdominal computed tomography and magnetic resonance imaging. In cases where a diagnosis cannot be established with imaging methods, diagnostic laparoscopy should be considered despite its invasive nature. Especially in female patients, diagnostic laparoscopy can be used both for diagnosis and treatment. We used abdominal ultrasonography and computed tomography for diagnostic purposes in the present case. Regression of lymphangiomas is unlikely. The recommended treatment is endoscopic polypectomy and mucosal resection for lesions smaller than 2 cm intraluminal, and surgical resection for lesions larger than 2 cm. (6,7,8,9) Due to the development of intussusception and acute abdomen in our case, we performed laparotomy and surgical resection. Postoperative examination revealed the cecal lymphangioma was 4 cm in size, making our treatment approach consistent with the literature. In the literature, the first colonic lymphangioma was described by Chisholm and Hillkowits (10). Only six cases of intussusception due to colonic lymphangioma have been reported in adults; five of them were located in the ascending colon and one was in the transverse colon. (11,12,13,14,15,16) As in our case, all six of those patients were female and had lesions over 4 cm in size, and all were treated with laparotomy and surgical resection. Lymphangioma, which is rare in adulthood, is generally detected by colonoscopy and endoscopic ultrasonography and treated when small. However, it must be kept in mind that in the rare cases where lymphangiomas reach large sizes, they may cause obstruction and intussusception and require treatment by surgical resection.

Ethics

Informed Consent: Consent form was filled out by all participants.

Peer-review: Internally peer-reviewed.

Conflict of Interest: No conflict of interest was declared by the author.

Financial Disclosure: The author declared that this study received no financial support.

References

(1.) de Perrot M, Rostan O, Morel P, Le Coultre C. Abdominal lymphangioma in adults and children. Br J Surg 1998;85:395-397.

(2.) Allen JG, Riall TS, Cameron JL, Askin FB, Hruban RH, Campbell KA. Abdominal lymphangiomas in adults. J Gastrointest Surg 2006;10:746-751.

(3.) Azar T, Berger DL. Adult intussusception. Ann Surg 1997;226:134-138.

(4.) Wegner G. Uber Lymphangioma. Arch Klin Chir 1877;20:641-707.

(5.) Matsuda T, Matsutani T, Tsuchia Y, Okhiama Y, Egami K, Yoshioka M, Maeda S, Onda M. A clinical evaluation of lymphangioma of large intestine: a case presentation of lymphangioma of the desending colon and a review of 279 Japanese cases. J Nippon Med Sch 2001;68:262-265.

(6.) Karasawa H, Miura T, Makino T, et al. Two cases of the large intestine extracted by endoscopic polypectomy. Prog Dig Endosc 1985;26:330-334.

(7.) Kurumato S, Sakai S, Tsuda K, Kaminishi M, Ihara O, Oohara T, Jinbo S, Murakami T. Lymphangioma of the large intestine. Report of a case. Dis Colon Rectum 1988;31:900-905.

(8.) Hatada T, Ikeda H, Tanigawa A, Fujiwara Y, Hanada Y, Yamamura T. Lymphangioma of the colon: a case report and review of the Japanese literature. Acta Gastroenterol Belg 2000;63:239-241.

(9.) Kohman ML, Wiersema MJ, Hawes RH, Canal D, Wiersema L. Preoperative diagnosis of cystic lymphangioma of the colon by endoscopic ultrasound. Gastrointest Endosc 1997;45:204-206.

(10.) Chisholm AJ, Hillkowits P. Lymphangioma of the rectum. Am J Surg 1932;17:281-282.

(11.) Nagle R. Lymphangiomatous hamartoma with intussusception of the caput caeci. Br J Surg 1968;55:879-880.

(12.) Matsui A, Okajima K, Ishii M, Namio H, Kawanishi M, Fujii Y, et al. Lymphangioma of the cecum with intussusceptions: a case report and review of literature. J Jpn Pract Surg Soc 1986;47:234-239.

(13.) Wan YL, Lee TY, Hung CF, Ng KK. Ultrasound and CT findings of a cecal lymphangioma presenting with intussusception. Eur J Radiol 1998;27:77-79.

(14.) Matsuba Y, Mizuiri H, Murata T, Niimi K. Adult intussusception due to lymphangioma of the colon. J Gastroenterol 2003;38:181-185.

(15.) Kim TO, Lee JH, Kim GH, Heo J, Kang DH, Song GA, Cho M. Adult intussusception caused by cystic lymphangioma of the colon: a rare case report. World J Gastroenterol 2006;12:2130-2132.

(16.) Kim DI, Seo HI, Kim JH, Kim HS, Jo HJ. Adult Intussusception due to Cecal Lymphangioma: a Case Report. J Korean Soc Coloproctol 2011;27:99-101.

[iD] Ecem Memisoglu

Hakkari State Hospital, Clinic of General Surgery, Hakkari, Turkey

Address for Correspondence/YazIsma Adresi: Ecem Memisoglu MD

Hakkari State Hospital, Clinic of General Surgery, Hakkari, Turkey

Phone: +90 533 721 52 52 E-mail: drecem@gmail.com ORCID ID: orcid.org/0000-0003-2360-4694

Received/Gelis Tarihi: 16.10.2017 Accepted/Kabul Tarihi: 29.11.2017

DOI: 10.4274/tjcd.44712
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Title Annotation:CASE REPORT
Author:Memisoglu, Ecem
Publication:Turkish Journal of Colorectal Disease
Article Type:Case study
Date:Jun 1, 2018
Words:1186
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