A Case of Cavernous Lymphangioma of the Small Bowel Mesentery

In Taik Hong; Jae Myung Cha; Joung Il Lee; Kwang Ro Joo; Il Hyun Baek; Hyun Phil Shin; Jung Won Jeon; Jun Uk Lim

doi:10.4166/kjg.2015.66.3.172

Journal List > Korean J Gastroenterol > v.66(3) > 1007420

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Hong, Cha, Lee, Joo, Baek, Shin, Jeon, and Lim: A Case of Cavernous Lymphangioma of the Small Bowel Mesentery

Case Report

Korean J Gastroenterol 2015;66(3):172-175.

Published online: 4 October 2015

DOI: https://doi.org/10.4166/kjg.2015.66.3.172

A Case of Cavernous Lymphangioma of the Small Bowel Mesentery

In Taik Hong, Jae Myung Cha, Joung Il Lee, Kwang Ro Joo, Il Hyun Baek, Hyun Phil Shin, Jung Won Jeon, Jun Uk Lim

Department of Internal Medicine, Kyung Hee University Hospital at Gangdong, Kyung Hee University School of Medicine, Seoul, Korea

Correspondence to: Jae Myung Cha, Department of Internal Medicine, Kyung Hee University Hospital at Gangdong, Kyung Hee University School of Medicine, 892 Dongnam-ro, Gangdong-gu, Seoul 05278, Korea. Tel: +82-2-440-6113, Fax: +82-2-440-6295, E-mail: drcha@khu.ac.kr

Received 11 February 2015 Revised 24 March 2015 Accepted 1 April 2015

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract

Lymphangioma is a benign vascular lesion with characteristics of subepithelial tumor which can proliferate in the lymphatic system. Lymphangioma of the small-bowel mesentery is rare, having been reported in less than 2% of all lymphangiomas. Lymphangioma does not require any specific treatment because it is absolutely a benign tumor. However, surgical exploration is rarely required for cases with disease-related symptoms or complications, or for those misdiagnosed as a malignant lesion. We recently experienced a case of mesenteric cavernous lymphangomas in a 53-year-old female who was misdiagnosed as having a liposarcoma. The final diagnosis was confirmed by a pathologic examination of the specimen that was obtained via laparoscopic exploration. Herein, we report a very rare case of mesenteric cavernous lymphangioma along with a brief review of relevant literature.

Keywords: Lymphangioma, Mesentery, Intestine, small, Neoplasms

References

1. Fonkalsrud EW. Congenital malformations of the lymphatic system. Semin Pediatr Surg. 1994; 3:62–69.

2. Tsukada H, Takaori K, Ishiguro S, Tsuda T, Ota S, Yamamoto T. Giant cystic lymphangioma of the small bowel mesentery: report of a case. Surg Today. 2002; 32:734–737.

3. Mosca RC, Pereira GA, Mantesso A. Cystic hygroma: characterization by computerized tomography. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2008; 105:e65–e69.

4. Hanagiri T, Baba M, Shimabukuro T, et al. Lymphangioma in the small intestine: report of a case and review of the Japanese literature. Surg Today. 1992; 22:363–367.

5. Na WT, Lee TH, Lee BS, et al. Clinical aspects of intraabdominal cystic lymphangioma in Korea. Korean J Gastroenterol. 2010; 56:353–358.

6. Batsakis JG. Tumors of the head and neck: clinical and pathological consideration. Volume 1. 2nd ed.Baltimore: Williams & Wilkins;1979. p. 301–302.

7. Chung JH, Suh YL, Park IA, et al. A pathologic study of abdominal lymphangiomas. J Korean Med Sci. 1999; 14:257–262.

8. Kahn HJ, Bailey D, Marks A. Monoclonal antibody D2–40, a new marker of lymphatic endothelium, reacts with Kaposi's sarcoma and a subset of angiosarcomas. Mod Pathol. 2002; 15:434–440.

9. Jeung MY, Gasser B, Gangi A, et al. Imaging of cystic masses of the mediastinum. Radiographics. 2002; 22(Suppl 1):S79–S93.

10. Vargas-Serrano B, Alegre-Bernal N, Cortina-Moreno B, Rodriguez- Romero R, Sanchez-Ortega F. Abdominal cystic lymphangiomas: US and CT findings. Eur J Radiol. 1995; 19:183–187.

11. Kim HH, Hur YH, Park CY, et al. Splenic cavernous lymphangioma mimicking splenic hemangioma. J Korean Surg Soc. 2009; 77:434–437.

12. Hwang SS, Choi HJ, Park SY. Cavernous mesenteric lymphangiomatosis mimicking metastasis in a patient with rectal cancer: a case report. World J Gastroenterol. 2009; 15:3947–3949.

13. Iyer R, Eftekhari F, Varma D, Jaffe N. Cystic retroperitoneal lymphangioma: CT, ultrasound and MR findings. Pediatr Radiol. 1993; 23:305–306.

14. Beneck D, Abati AD, Greco MA. Lymphangioma presenting as a nasal polyp in an infant. Arch Pathol Lab Med. 1985; 109:773–775.

15. Price C. Cystic lymphangioma in the maxillary sinus: a case report. Br J Oral Surg. 1976; 14:41–46.

16. Chung WC, Kim HK, Yoo JY, et al. Colonic lymphangiomatosis associated with anemia. World J Gastroenterol. 2008; 14:5760–5762.

17. Lee JH, Lee SY, Lee JH, et al. A case of colonic intussusception caused by cystic lymphangioma of the colon. Korean J Gastrointest Endosc. 2006; 32:226–230.

18. Ha CY, Shim KN, Kang MJ, et al. A case of cystic lymphangioma of small bowel mesentery presented with small bowel volvulus. Intest Res. 2008; 6:76–79.

19. Saba C, Bossi MC, Barletta A. Therapy of subcutaneous cystic lymphangioma with ultrasound-guided puncture and alcoholi-zation. Radiol Med. 1992; 83:270–272.

20. Okada A, Kubota A, Fukuzawa M, Imura K, Kamata S. Injection of bleomycin as a primary therapy of cystic lymphangioma. J Pediatr Surg. 1992; 27:440–443.

Fig. 1.

Abdominal CT findings. (A) Initial CT shows 1.3 cm sized soft tissue mass (arrow) in small bowel mesentery. (B) Follow-up CT shows increased size of the lesions from 1.3 to 2 cm (arrowheads) compared with previous CT scan performed 7 months ago.

Fig. 2.

Laparoscopic exploration demonstrates mesenteric fat thickening and cystic mass.

Fig. 3.

Gross finding of resected specimen. Gross pathologic examination reveals a 3.5×1.5×1.2 cm sized mass composed of various- sized cysts.

Fig. 4.

Microscopic findings. (A) There are many dilated lymphatic vessels lined by a single layer of endothelium (H&E, ×40). (B) Dilated lymphatic vessels are filled with proteinaceous fluid (H&E, ×100). (C) The lining cells are positive for D2–40 (immunohistochemical stain, ×100).

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