Cyanoacrylate glue in the management of blue rubber bleb nevus syndrome

Case 1. A 16-year-old boy with blue rubber bleb nevus syndrome (BRBNS) was admitted for occult bleeding. He had a history of gastric and colonic hemangiomas treated 2 years earlier by argon plasma coagulation and alcohol injection respectively. Clinical examination showed a few hemangiomas on his back and feet ([Fig. 1]).

Fig. 1 Case 1. Hemangiomas of the foot.

Capsule endoscopy (Pillcam SB2; Given Imaging, Yoqneam, Israel) disclosed three blebs of the small bowel ([Fig. 2]), and the patient underwent double-balloon enteroscopy (Fujinon, Saitama, Japan).

Fig. 2 Case 1. Capsule endoscopy picture of a bluish, nipple-like rubbery vascular lesion of the jejunum, compatible with a blue rubber bleb nevus.

A 2-cm bleb was found at the jejunum, two smaller lesions at the ileum, and one at the transverse colon. N-butyl-2-cyanoacrylate and methacryloxysulfolane (Glubran 2), 1 ml, diluted with Lipiodol, 1 ml, was injected in aliquots of 1 ml per injection and up to 4 ml at each lesion ([Fig. 3]). No complications occurred. At 6-month follow-up the patient had no anemia.

Fig. 3 a Case 1. Hemangioma of the proximal jejunum. b Solidification of the glue inside the lumen. Inadvertent sticking of the needle inside the lesion or adherence of the glue to the needle was avoided by flushing at least 2 ml water for injection after each aliquot of Glubran 2 and Lipiodol. c Typical lesion of the transverse colon.

Case 2. An 11-year-old girl with BRBNS was addressed for hematochezia due to colonic blebs. Colonoscopy revealed a bleeding lesion at the cecum ([Fig. 4]).

Fig. 4 Case 2. Bleeding hemangioma of the cecum near the ileocecal valve.

After initial failure to stop the bleeding with a hemostatic grasper (Coagrasper, Olympus), Glubran 2 was injected successfully. Two more blebs of the right colon were treated similarly. After 2 years the patient remained asymptomatic.

BRBNS is a rare condition characterized by multiorgan cavernous hemangiomas. Gastrointestinal bleeding is the major problem, and several endoscopic therapeutic approaches have been proposed: Nd:YAG laser, bipolar or argon plasma coagulation, band ligation, snare resection, and sclerotherapy [1] [2] [3] [4]. Our patients were treated successfully by Glubran 2 injection, commonly used in the management of gastric varices. Endoscopic obliteration of cavernous hemangiomas with n-butyl-2-cyanoacrylate (Histoacryl) has been previously described, in the rectum [5]. However, these appear to be the first cases of cyanoacrylate glue injection to control bleeding in the small bowel and colon in the setting of BRBNS.

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References

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• 2 Maunoury V, Turck D, Brunetaud J M et al. Blue rubber bleb nevus syndrome. 3 cases treated with a Nd:YAG laser and bipolar electrocoagulation [in French with English abstract].  Gastroenterol Clin Biol. 1990;  14 593-595
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• 5 Zurakowski J, Swiercz P, Wróblewski T et al. Diffuse cavernous hemangioma of rectosigmoid colon treated with n-butyl-2-cyanoacrylate injections.  Endoscopy. 2008;  40 E120-121