Skip to main page content
U.S. flag

An official website of the United States government

Dot gov

The .gov means it’s official.
Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

Https

The site is secure.
The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
Case Reports
. 2012 Nov;83(5):316-20.
doi: 10.4174/jkss.2012.83.5.316. Epub 2012 Oct 29.

Radical resection of intestinal blue rubber bleb nevus syndrome

Kang Kook Choi  1 , Jin Yong Kim, Min Jung Kim, Hyojun Park, Dong Wook Choi, Seong Ho Choi, Jin Seok Heo
Affiliations
Case Reports

Radical resection of intestinal blue rubber bleb nevus syndrome

Kang Kook Choi et al. J Korean Surg Soc. 2012 Nov.

Abstract

Blue rubber bleb nevus syndrome (BRBNS) is a rare systemic vascular disorder characterized by multiple venous malformations involving many organs. BRBNS can occur in various organs, but the most frequently involved organs are the skin and gastrointestinal (GI) tract. GI lesions of BRBNS can cause acute or chronic bleeding, and treatment is challenging. Herein, we report a case of GI BRBNS that was successfully treated with a combination of intraoperative endoscopy and radical resection.

Keywords: Bleeding; Blue rubber bleb nevus syndrome; Hemorrhage; Intestines.

PubMed Disclaimer

Conflict of interest statement

No potential conflict of interest relevant to this article was reported.

Figures

Fig. 1
Fig. 1
A 0.5 cm hemangioma was found in the sigmoid colon without evidence of bleeding on colonoscopy.
Fig. 2
Fig. 2
The small intestine was explored to the proximal jejunum, and three hemangiomas were found. Two hemangiomas had mucosal erosions. (A) The most proximal hemangioma was located in the duodenojejunal junction. (B) The middle hemangioma. (C) The most distal hemangioma was found in the proximal jejunum.
Fig. 3
Fig. 3
(A) The tattoo performed via double-balloon endoscopy was observed. (B, C) Four other hemangiomas were identified distal to the tattooed point of the intestine. (D) Segmental resection and end-to-end anastomosis were performed for two segments that had two hemangiomas in close proximity.
See this image and copyright information in PMC

References

    1. Domini M, Aquino A, Fakhro A, Tursini S, Marino N, Di Matteo S, et al. Blue rubber bleb nevus syndrome and gastrointestinal haemorrhage: which treatment? Eur J Pediatr Surg. 2002;12:129–133. - PubMed
    1. Hasosah MY, Abdul-Wahab AA, Bin-Yahab SA, Al-Rabeaah AA, Rimawi MM, Eyoni YA, et al. Blue rubber bleb nevus syndrome: extensive small bowel vascular lesions responsible for gastrointestinal bleeding. J Paediatr Child Health. 2010;46:63–65. - PubMed
    1. Shepherd V, Godbolt A, Casey T. Maffucci's syndrome with extensive gastrointestinal involvement. Australas J Dermatol. 2005;46:33–37. - PubMed
    1. Shovlin CL, Guttmacher AE, Buscarini E, Faughnan ME, Hyland RH, Westermann CJ, et al. Diagnostic criteria for hereditary hemorrhagic telangiectasia (Rendu-Osler-Weber syndrome) Am J Med Genet. 2000;91:66–67. - PubMed
    1. Deng ZH, Xu CD, Chen SN. Diagnosis and treatment of blue rubber bleb nevus syndrome in children. World J Pediatr. 2008;4:70–73. - PubMed

Publication types