Case Report

Cavernous Hemangioma of Colon and Pregnancy: A Case Report

Abstract

Cavernous hemangioma of the colon is a rare vascular malformation but is clinically important because it can sometimes cause massive bleeding. We report a case of thrombocytopenia in a post-partum young woman with large cavernous hemangioma of the ileum and colon, culminating in Kasabach Merritt syndrome (KMS), massive uncontrollable hemorrhage, and death. CT scan shows multiple submucosal bulgings in all segments of the colon. Macroscopic examination showed multiple well-defined masses, with gray-brown color in ileum, cecum and ascending colon. The masses were found to be a cavernous hemangioma with a thick fibrotic wall and extensive intra-cystic hemorrhage. KMS is an uncommon complication of colonic large hemangiomas that, as in our patient, can lead to uncontrollable bleeding and death and should be kept in mind by visceral surgeons as one differential diagnosis of large intra-abdominal tumorous masses, especially in young adults.

Hang He, Zunguo Du, Sijie Hao, Lie Yao, Feng Yang, Yang Di, Ji Li, Yongjian Jiang, Chen Jin and Deliang Fu. Adult primary retroperitoneal cavernous hemangioma: a case report, World Journal of Surgical Oncology 2012;10:261.

Yoo S. GI-Associated Hemangiomas and Vascular Malformations. Clinics in Colon and Rectal Surgery. 2011;24(3):193-200. doi:10.1055/s-0031-1286003.

Pontecorvo C, Lombardi S, Mottola L, Donisi M. DiTuoro A. Hemangiomas of the large bowel. Report of a case. Dis Colon Rectum 1983; 26:818–820.

Vilallonga, R., Espin Basany, E., Armengol, M. Cavernous hemangioma: Unusual benign tumor of the transverse colon, Turk J Gastroenterol 2009; 20(2):146-149.

van Deursen CT, Buijs J, Nap M. An uncommon polyp in the colon: a pedunculated cavernous hemangioma. Endoscopy 2008; 40 Suppl 2: E127.

Kimura S, Tanaka S, Kusunoki H, Kitadai Y, Sumii M, Tazuma S, Yoshihara M, Haruma K, Chayama K. Cavernous hemangioma in the ascending colon treated by endoscopic mucosal resection. J Gastroenterol Hepatol 2007; 22:280–281.

Hasegawa K, Lee WY, Noguchi T, Yaguchi T, Sasaki H, Nagasako K. Colonoscopic removal of hemangiomas. Dis Colon Rectum 1981; 24:85–89.

Marcelo AF, Ribeiro Jr, Francine P, Juliana M Gonçalves, and Eleazar C. Spontaneous rupture of hepatic hemangiomas: A review of the literature, World J Hepatol 2010; 2(12):428–433.

Sylla P, Deutsch G, Luo J, Recavarren C, Kim S, Heimann TM, Steinhagen RM. 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 2008; 23:653–658.

Marinis A, Kairi E, Theodosopoulos T, Kondi-Pafiti A, Smyrniotis V. Right colon and liver hemangiomatosis: a case report and a review of the literature. World J Gastroenterol 2006; 12:6405–6407.

Head HD, Baker JQ, Muir RW. Hemangioma of the colon.Am J Surg 1973; 126:691–694.

Lyon DT, Mantia AG. Large-bowel hemangiomas. Dis Colon Rectum 1984; 27:404-414.

Levy AD, Abbott RM, Rohrmann CA Jr, Frazier AA, Kende A. Gastrointestinal hemangiomas: imaging findings with pathologic correlation in pediatric and adult patients. AJR Am J Roentgenol 2001; 177:1073–1081.

Hsu RM, Horton KM, Fishman EK. Diffuse cavernous hemangiomatosis of the colon: findings on three-dimensional CT colonography. AJR Am J Roentgenol 2002; 179:1042-1044.

Geenen RWF, den Bakker MA, Bangma CH, Hussain SM, Krestin GR. Sonography CT MRI of giant cavernous hemangioma of the kidney: correlation with pathologic findings. Am J Roentgenol 2004; 182:411-414.

Zhao XK, Zhang JQ, Zhong ZH, Koh CJ, Xie HW, Hardy BE. Large renal cavernous hemangioma with renal vein thrombosis: case report and review of literature 2009.

Yoo S. GI-Associated Hemangiomas and Vascular Malformations. Clinics in Colon and Rectal Surgery. 2011;24(3):193-200. doi:10.1055/s-0031-1286003.

Sarkar, M., Mulliken, J. B.; Kozakewich, H. P. W.; Robertson, R. L.; Burrows, P. E. Thrombocytopenic Coagulopathy (Kasabach-Merritt Phenomenon) Is Associated with Kaposiform Hemangioendothelioma and Not with Common Infantile Hemangioma, Plastic & Reconstructive Surgery 1997; 100(6):1377.

Amarapurkar D, Jadliwala M, Punamiya S, Jhawer P, Chitale A, Amarapukar A. Cavernous hemangiomas of the rectum: report of three cases. Am J Gastroenterol 1998; 93:1357-1359.

Yeoman LJ, Shaw D. Computerized tomography appearances of pelvic haemangioma involving the large bowel in childhood. Pediatr Radiol 1989; 19:414-416.

Sharma S, Gandhi D, Thulkar S, Dwivedi DN. Role of computed tomography in diagnosis of diffuse cavernous hemangioma of the rectum. Am J Gastroenterol 1999; 94:1986-1987.

Angela D. Levy, Robert M. Abbott, Charles A. Rohrmann, Jr. Gastrointestinal hemangiomas: Imaging findings with pathologic correlation in pediatric and adult patients. AJR Am J Roentgenol 2001; 177:10731081.

Bortz JH. Diffuse cavernous hemangioma of the rectum and sigmoid. Abdom Imaging 1994; 19:18-20.

Perez C, Andreu J, Llauger J, Valls J. Hemangioma of the rectum: CT appearance. Gastrointest Radiol 1987; 12:347-349.

Mundinger GS, Gust S, Micchelli ST, Fishman EK, Hruban RH, Wolfgang CL. Adult pancreatic hemangioma: case report and literature review. Gastroenterol Res Pract 2009; 839730.

Chang WT, Lee KT, Yang SF. Cavernous hemangioma of the pancreas: report of a case. Pancreas 2003; 26:310-312.

De Pree, M. J. A Case of Giant Hemangioma with Thrombocytopenia 1963; DOI: 10.1111/j.1651-2227.1963.tb03799.

Ryu, J., Park, K. S., Il-Young Cheong, II-Y., Kim, D. K., Do, S.-H., Kim, J. K. Rupture of hepatic hemangioma during pregnancy−A case report, Anesth Pain Med 2008; 3:313-315.

Gottlieb, K., Coff, P., Preiksaitis, H., Juviler, A., Fern, P. Massive Hemorrhage in Pregnancy Caused by a Diffuse Cavernous Hemangioma of the Rectum – EUS as Imaging Modality of Choice, Medscape J Med 2008; 10(9):206.

Chatzoulis G, Kaltsas A, Daliakopoulos S, Sallam O, Maria K, Chatzoulis K, Pachiadakis I. Co-existence of a giant splenic hemangioma and multiple hepatic hemangiomas and the potential association with the use of oral contraceptives: a case report. J Med Case Reports 2008; 2:147.

Xiao X, Liu J, Sheng M. Synergistic effect of estrogen and VEGF on the proliferation of hemangioma vascular endothelial cells. J Pediatr Surg 2004; 39:1107–1110.

Takayoshi K., Massanobu T. polpoid cavernous hemangioma removed under colonoscopy, Digetive Endoscopy 2003; 15:338-340.

Lei H, Sui B, Liu D: Comprehensive sequential therapy of kasabach-merritt syndrome. Nat Med J China. 2011, 91:2538-2541.

Yoon HS, Lee JH, Moon HN, Seo JJ, Im HJ, Goo HW: Successful treatment of retroperitoneal infantile hemangioendothelioma with kasabach-merritt syndrome using steroid, α-interferon, and vincristine. J Pediatr Hematol Oncol 2009; 31:952-954. 10.1097/MPH.0b013e3181b7eb24.

Hauer J, Graubner U, Konstantopoulos N, Schmidt S, Pfluger T, Schmid I: Effective treatment of kaposiform hemangioendotheliomas associated with kasabach-merritt phenomenon using four-drug regimen. Pediatr Blood Cancer 2007; 49:852-854. 10.1002/pbc.20750.

Azad, T., Hasan, R., Bhattacherjee, A. and Choudhury, T. 2012. A case report on Kasabach Merrit Syndrome, Bangladesh J. Child Health 2012; 38(2):105-107.

Arunachalam, P., Kumar, V.R. and Swathi, D., Kasabach-Merritt syndrome with large cutaneous vascular tumors. Journal of Indian Association of Pediatric Surgeons 2012; 17(1):33-36.

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IssueVol 56, No 4 (2018) QRcode
SectionCase Report(s)
Keywords
Cavernous hemangioma Colon Kasabach-meritt syndrome

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How to Cite
1.
Zare Mehrjardi A, Fakhari S, Alem Rajab M. Cavernous Hemangioma of Colon and Pregnancy: A Case Report. Acta Med Iran. 2018;56(4):272-277.