Multiendoscope-assisted treatment for blue rubber bleb
nevus syndrome

Watanabe, Yuji; Sato, Mariko; Tokui, Kazuyoshi; Yukumi, Shungo; Koga, Shigehiro; Nezu, Kenji; Matsui, Hidetaka; Murakami, Hidehiro; Kawachi, Kanji

doi:10.1007/s004640000095

Cited by 8 publications

(4 citation statements)

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“…Panendoscopy, push enteroscopy, and colonoscopy provide the diagnostic, prognostic, and therapeutic elements to improve patient outcomes and to reduce morbidity and mortality from severe gastrointestinal hemorrhage. 9,[11][12][13][14] Domini et al 9 reported that management with electrocautery or laser photocoagulation were usually not effective, although some reports recommend them. [15][16][17][18] The influence of pregnancy on the prognosis of BRBNS is unknown because only few reports exist.…”

Section: Discussionmentioning

confidence: 99%

Severe Gastrointestinal Bleeding during Pregnancy in a Case of Blue Rubber Bleb Nevus Syndrome

Kanai¹,

Noike²,

Masaki³

et al. 2005

Semin Thromb Hemost

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Blue rubber bleb nevus syndrome (BRBNS) is a rare disorder characterized by distinctive cutaneous and gastrointestinal venous malformations that usually cause massive or occult gastrointestinal hemorrhage and iron deficiency anemia secondary to the bleeding episodes. Only two reports of BRBNS in pregnant women can be found, according to the MEDLINE search. Both cases did not describe severe complications during pregnancy. This is the first case report of BRBNS with severe intestinal hemorrhage in a pregnant woman. The woman had been symptom-free for 5 years before the pregnancy and remained symptom-free for 2 years after delivery. This suggests that pregnancy induced the hemangiomas of the gastrointestinal tract to bleed. Celiotomy was needed to deal with massive gastrointestinal bleeding. We propose that women with BRBNS should be checked for systemic hemangiomas before pregnancy. However, it is difficult to check the small intestine thoroughly for hemangioma. Therefore, adequate attention needs to be paid to possible gastrointestinal bleeding when caring for patients with this condition. Our experience suggests that when dealing with pregnant women with BRBNS, it is essential to bear in mind the possibility that pregnancy may trigger the symptoms of hemangioma to become manifest, possibly causing life-threatening massive bleeding from the digestive tract.

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Section: Discussionmentioning

confidence: 99%

Severe Gastrointestinal Bleeding during Pregnancy in a Case of Blue Rubber Bleb Nevus Syndrome

Kanai¹,

Noike²,

Masaki³

et al. 2005

Semin Thromb Hemost

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“…[6] The association of this condition with congenital heart disease is very rare with only a single case report so far. [7] To the best of our knowledge, this is the first report of a case documenting BRBNS associated with tetralogy of Fallot.…”

Section: Discussionmentioning

confidence: 75%

Unusual Cause and Association of Gastrointestinal Bleed in a Young Boy

Kanojia

et al. 2017

Journal of Digestive Endoscopy

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A 4-year old child, previously operated case of tetralogy of Fallot present with recurrent episodes of massive lower gastrointestinal bleed of one year duration. Endoscopic evaluation revealed multiple bluish vascular lesions in the duodenum and proximal jejunum and a single rectal polyp. Histology of the lesion was suggestive of venous malformation consistent with blue rubber bleb nevus syndrome (BRBNS). Child underwent endoscopic snaring and surgical resection with end to end anastomosis. At six months’ follow-up child was asymptomatic without any bleed episodes.

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“…Bowel resection is reserved for localised lesions, significant bleeding that cannot be controlled suitably by endoscopy or radiology, and for complications such as ischaemia and intussusception [ 21 ]. Role of elective radical surgical excision remains debatable [ 22 , 23 ]. Anti-angiogenic agents including corticosteroids, interferon α therapy and gamma globulin have been attempted [ 24 , 25 ].…”

Section: Discussionmentioning

confidence: 99%