Literature DB >> 25631979

Blue rubber bleb nevus syndrome: a rare cause of chronic gastrointestinal bleed in adults.

Gayatri Amit Deshpande1, Inian Samarasam, Sam Varghese George, Sudhakar Chandran.   

Abstract

Blue rubber bleb nevus syndrome (BRBNS) is a rare condition characterised by venous malformations in the skin, gastrointestinal tract and other parts of the body. Its presentation is usually sporadic, although cases of autosomal dominant inheritance have been reported. Usually seen in children, BRBNS presentation in adults is rare. Symptoms at presentation depend on the organs involved; patients with BRBNS may present with acute or chronic gastrointestinal bleed. We herein report a rare presentation of BRBNS in an adult who suffered from intermittent abdominal pain and melaena for three years. Contrast-enhanced computed tomography revealed a jejunojejunal intussusception with a vascular malformation as the lead point. The patient underwent laparotomy with resection of the intussuscepted bowel segment. Recovery was uneventful. In spite of a wide range of therapeutic options for the management of BRBNS described in the literature, the efficacy of those available therapies, including surgical excision, is not well established.

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Year:  2014        PMID: 25631979      PMCID: PMC4294001          DOI: 10.11622/smedj.2014164

Source DB:  PubMed          Journal:  Singapore Med J        ISSN: 0037-5675            Impact factor:   1.858


  9 in total

Review 1.  Blue rubber bleb nevus syndrome causing upper GI hemorrhage: a novel management approach and review.

Authors:  Manisha Dwivedi; Sri Prakash Misra
Journal:  Gastrointest Endosc       Date:  2002-06       Impact factor: 9.427

2.  Blue rubber bleb nevus syndrome: surgical eradication of gastrointestinal bleeding.

Authors:  Steven J Fishman; C Jason Smithers; Judah Folkman; Dennis P Lund; Patricia E Burrows; John B Mulliken; Victor L Fox
Journal:  Ann Surg       Date:  2005-03       Impact factor: 12.969

Review 3.  Combined laser-steroid therapy in blue rubber bleb nevus syndrome: case report and review of the literature.

Authors:  K Dieckmann; C Maurage; N Faure; A Margulies; G Lorette; J Rudler; J C Rolland
Journal:  Eur J Pediatr Surg       Date:  1994-12       Impact factor: 2.191

4.  Blue rubber bleb nevus (Bean syndrome): evolution of four cases and clinical response to pharmacologic agents.

Authors:  M D Boente; M R Cordisco; M D Frontini; R A Asial
Journal:  Pediatr Dermatol       Date:  1999 May-Jun       Impact factor: 1.588

5.  Chronic subcutaneous octreotide decreases gastrointestinal blood loss in blue rubber-bleb nevus syndrome.

Authors:  D Gonzalez; B J Elizondo; S Haslag; G Buchanan; J S Burdick; P C Guzzetta; B A Hicks; J M Andersen
Journal:  J Pediatr Gastroenterol Nutr       Date:  2001-08       Impact factor: 2.839

6.  Blue rubber bleb nevus syndrome with disseminated intravascular coagulation and thrombocytopenia: successful treatment with high-dose intravenous gammaglobulin.

Authors:  M Aihara; Y Konuma; K Okawa; R Komai; I Kudo; R Morioka; K Kariya; H Takami; Y Sawada; A Munakata
Journal:  Tohoku J Exp Med       Date:  1991-02       Impact factor: 1.848

7.  Blue rubber bleb nevus syndrome presenting with recurrences.

Authors:  K S Sandhu; H Cohen; R Radin; F S Buck
Journal:  Dig Dis Sci       Date:  1987-02       Impact factor: 3.199

8.  A gene for familial venous malformations maps to chromosome 9p in a second large kindred.

Authors:  C J Gallione; K A Pasyk; L M Boon; F Lennon; D W Johnson; E A Helmbold; D S Markel; M Vikkula; J B Mulliken; M L Warman
Journal:  J Med Genet       Date:  1995-03       Impact factor: 6.318

9.  Synchronous multiple small bowel intussusceptions in an adult with blue rubber bleb naevus syndrome: Report of a case and review of literature.

Authors:  Clement Lee; Debasish Debnath; Tara Whitburn; Mark Farrugia; Federico Gonzalez
Journal:  World J Emerg Surg       Date:  2008-01-18       Impact factor: 5.469

  9 in total
  3 in total

1.  Sirolimus as a promising drug therapy for blue rubber bleb nevus syndrome: Two-case report.

Authors:  Jian-Xun Ma; You-Chen Xia; Li-Ping Zou; Heng-Ju Lin; Xu Chang; Li-Ying Liu; Joy Roechelle A Toledo
Journal:  SAGE Open Med Case Rep       Date:  2022-05-11

2.  Blue rubber bleb nevus syndrome: our experience and new endoscopic management.

Authors:  Wenguo Chen; Hongtan Chen; Guodong Shan; Ming Yang; Fengling Hu; Qi Li; Lihua Chen; Guoqiang Xu
Journal:  Medicine (Baltimore)       Date:  2017-08       Impact factor: 1.889

Review 3.  Blue rubber bleb nevus syndrome with the complication of intussusception: A case report and literature review.

Authors:  Zeming Hu; Xuan Lin; Jianing Zhong; Qingfang He; Qin Peng; Jianbo Xiao; Bin Chen; Jianhong Zhang
Journal:  Medicine (Baltimore)       Date:  2020-07-10       Impact factor: 1.817

  3 in total

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