Korean J Gastroenterol.  2023 Aug;82(2):91-95. 10.4166/kjg.2023.057.

Cystic Lymphangioma of Rectum-A Case Report and Review of Literature

• Dessai GS
• Soni NK
• Pujari S
• Prabhu R
• Kantharia CV

• KMID: 2545333
• DOI: http://doi.org/10.4166/kjg.2023.057

Abstract

Colorectal cystic lymphangiomas are rare benign lesions. They are characterized by the presence of either single or multi-cystic spaces lined by endothelium. Though there are multiple case reports of right and transverse colonic lymphangioma; only around 10 cases of lymphangioma of the rectum have been reported. We present a case report of rectal lymphangioma with the relevant literature review.

Keyword

Lymphangioma; Cystic lymphangioma; Rectum

Figure

• 

  Fig. 1 Contrast enhanced computed tomography is suggestive of a multiloculated cystic lesion with well-defined enhancing walls arising from the upper rectum (axial sections). The black arrows indicate the loss of the fat plane between the lymphangioma and rectal wall (original images)

• 

  Fig. 2 Contrast enhanced computed tomography-sagittal section demonstrating the relation of lymphangioma to the urinary bladder.

• 

  Fig. 3 Colonoscopy was normal.

• 

  Fig. 4 Resected low anterior resection specimen with a cystic lesion along the right rectal wall (original image).

• 

  Fig. 5 Gross specimen after resection suggests normal mucosa with submucosal cystic spaces with extramural extension.

• 

  Fig. 6 Histopathological examination (HPE) suggestive of multiple cystic spaces filled with lymph lined by endothelium and separated by fibrous septae in the submucosa of the rectum with an extraserosal component. Hematoxylin and eosin (H&E) stain with 20× magnification (original image).

Reference

1. Chisholm AJ, Hillkowits P. 1932; Lymphangioma of the rectum. Am J Surg. 17:281–282. DOI: 10.1016/S0002-9610(32)90497-8.

2. Matsuda T, Matsutani T, Tsuchiya Y, et al. 2001; A clinical evaluation of lymphangioma of the large intestine: a case presentation of lymphangioma of the descending colon and a review of 279 Japanese cases. J Nippon Med Sch. 68:262–265. DOI: 10.1272/jnms.68.262. PMID: 11404774.

3. Sylla P, Deutsch G, Luo J, et al. 2008; Cavernous, arteriovenous, and mixed hemangioma-lymphangioma of the rectosigmoid: rare causes of rectal bleeding--case series and review of the literature. Int J Colorectal Dis. 23:653–658. DOI: 10.1007/s00384-008-0466-4. PMID: 18330577.

4. Pandey S, Fan M, Zhu J, Lu X, Chang D, Li X. 2017; Unusual cause of 55 years of rectal bleeding: hemolymphangioma (a case report). Medicine (Baltimore). 96:e6264. DOI: 10.1097/MD.0000000000006264. PMID: 28272235. PMCID: PMC5348183.

5. Zagaceta Torres W, Ramírez García JF, Chá Vez Rosell MÁN. 2020; [Cystic lymphangioma of the rectum-sigmoid in a public hospital in Lima-Peru: case report]. Rev Gastroenterol Peru. 40:64–68. Spanish. DOI: 10.47892/rgp.2020.401.1032. PMID: 32369468.

6. Kumar S, Sarkar D, Prasad S, et al. 2012; Large pelvic masses of obscure origin: urologist's perspective. Urol Int. 88:215–224. DOI: 10.1159/000334332. PMID: 22377534.

7. Chen G, Cui W, Ji XQ, Du JF. 2013; Diffuse hemolymphangioma of the rectum: a report of a rare case. World J Gastroenterol. 19:1494–1497. DOI: 10.3748/wjg.v19.i9.1494. PMID: 23538679. PMCID: PMC3602512.

8. Kim KM, Choi KY, Lee A, Kim BK. 2000; Lymphangioma of large intestine: report of ten cases with endoscopic and pathologic correlation. Gastrointest Endosc. 52:255–259. DOI: 10.1067/mge.2000.107710. PMID: 10922105.

9. Matsuba Y, Mizuiri H, Murata T, Niimi K. 2003; Adult intussusception due to lymphangioma of the colon. J Gastroenterol. 38:181–185. DOI: 10.1007/s005350300030. PMID: 12640534.

10. Chen CF, Chuang CH, Lu CY, Hu C, Kuo TL, Hsieh JS. 2009; Adult intussusception secondary to lymphangioma of the cecum: a case report. Kaohsiung J Med Sci. 25:347–352. DOI: 10.1016/S1607-551X(09)70527-3. PMID: 19561001.

11. Ly MMG, De Robles MS, Mckenzie C, Young CJ. 2019; Colonic lymphangioma presenting with intermittent pain and intussusception. J Surg Case Rep. 2019:rjy336. DOI: 10.1093/jscr/rjy336. PMID: 30651966. PMCID: PMC6329364.

12. Kim DI, Seo HI, Kim JH, Kim HS, Jo HJ. 2011; Adult intussusception due to cecal lymphangioma: A case report. J Korean Soc Coloproctol. 27:99–101. DOI: 10.3393/jksc.2011.27.2.99. PMID: 21602970. PMCID: PMC3092083.

13. Kim TO, Lee JH, Kim GH, et al. 2006; Adult intussusception caused by cystic lymphangioma of the colon: a rare case report. World J Gastroenterol. 12:2130–2132. DOI: 10.3748/wjg.v12.i13.2130. PMID: 16610070. PMCID: PMC4087698.

14. Kunimura T, Inagaki T, Hayashi R, Katou H, Iwaku K, Morohoshi T. 2005; A case of multiple colonic lymphangiomas causing intussusception. J Gastroenterol. 40:316–318. DOI: 10.1007/s00535-004-1544-1. PMID: 15830294.

15. Sugiura H, Katoh H, Inomata H, Maeda Y, Saitoh M, Tanabe T. 1990; A case of bleeding lymphangioma of the large intestine. J Surg Hokkaido. 35:123–126.

16. Zilko PJ, Laurence BH, Sheiner H, Pollard J. 1975; Cystic lymphangiomyoma of the colon causing protein-losing enteropathy. Am J Dig Dis. 20:1076–1080. DOI: 10.1007/BF01071198. PMID: 1200000.

17. Morozumi K, Omoto T, Miyazaki H, et al. 1999; Two cases of lymphangioma of the colon accompanied with colon cancer. Gastroenterol Endosc. 41:1323–1329.

18. Young TH, Ho AS, Tang HS, Hsu CT, Lee HS, Chao YC. 1996; Cystic lymphangioma of the transverse colon: report of a case and review of the literature. Abdom Imaging. 21:415–417. DOI: 10.1007/s002619900094. PMID: 8832861.

19. Bhutani MS, Annangi S, Koduru P, Aggarwal A, Suzuki R. 2016; Diagnosis of cystic lymphangioma of the colon by endoscopic ultrasound: Biopsy is not needed! Endosc Ultrasound. 5:335–338. DOI: 10.4103/2303-9027.191668. PMID: 27803907. PMCID: PMC5070292.

20. Zhuo CH, Shi DB, Ying MG, et al. 2014; Laparoscopic segmental colectomy for colonic lymphangiomas: a definitive, minimally invasive surgical option. World J Gastroenterol. 20:8745–8750. DOI: 10.3748/wjg.v20.i26.8745. PMID: 25024636. PMCID: PMC4093731.

21. Wegner G. 1877; Uber lymphangiome. Arch Klin Chir. 20:641–707.

22. Wiegand S, Eivazi B, Barth PJ, et al. 2008; Pathogenesis of lymphangiomas. Virchows Arch. 453:1–8. DOI: 10.1007/s00428-008-0611-z. PMID: 18500536.