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.
Endoscopy_UCTN_Code_TTT_1AO_2AD
