J Korean Surg Soc.  2012 Nov;83(5):316-320. 10.4174/jkss.2012.83.5.316.

Radical resection of intestinal blue rubber bleb nevus syndrome

• Choi KK
• Kim JY
• Kim MJ
• Park H
• Choi DW
• Choi SH
• Heo JS

• KMID: 2145009
• DOI: http://doi.org/10.4174/jkss.2012.83.5.316

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.

Keyword

Intestines; Blue rubber bleb nevus syndrome; Bleeding; Hemorrhage

MeSH Terms

Blister
Endoscopy
Gastrointestinal Neoplasms
Hemorrhage
Intestines
Nevus
Nevus, Blue
Rubber
Skin
Skin Neoplasms
Gastrointestinal Neoplasms
Nevus, Blue
Rubber
Skin Neoplasms

Figure

• 

  Fig. 1 A 0.5 cm hemangioma was found in the sigmoid colon without evidence of bleeding on colonoscopy.

• 

  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 (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.

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