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Blue Rubber Bleb Nevus Syndrome as a Cause of Lower Digestive Bleeding

DOI: 10.1155/2014/683684

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Abstract:

Introduction. Blue rubber bleb nevus syndrome is a rare disorder that is characterized by multiple recurrent vascular malformations that involve the skin and the gastrointestinal tract. The disease can present chronic anemia and severe episodes of gastrointestinal bleeding. Case Report. A 41-year-old man was admitted with recurrent episodes of lower gastrointestinal bleeding and anemia that had worsened over the last 3 months. The physical examination showed soft, diffuse, compressible, bluish nodules on all of the skin surfaces of the body. A biopsy from one of these skin lesions allowed a histological diagnosis of cavernous hemangioma. He submitted to a colonoscopy, which showed hemorrhoids and a plane vascular lesion mainly located on the right colon with recent signs of bleeding; this lesion was treated by local excision and sclerosis. The pathological study of the colon specimens also reflected the presence of cavernous hemangioma. The cutaneous hemangiomas and the presence of colonic venous malformations were compatible with blue rubber bleb nevus syndrome. The patient presented a favorable follow-up with clinical control of the anemia and without relapse of the gastrointestinal bleeding two years after the procedure. Conclusion. Although rarely diagnosed, blue rubber bleb nevus syndrome may be responsible for lower digestive bleeding. 1. Introduction Blue rubber nevus syndrome (BRNS), also known as Bean Syndrome, is a rare angiomatosis condition with an estimated incidence of 1?:?14,000 births [1, 2]. This syndrome is characterized by the association of cutaneous and gastrointestinal venous malformations with iron deficiency anemia or gastrointestinal bleeding [3]. The disease was described by Gascoyen (1860) [4], but Bean differentiated this type of angiomatosis from other vascular diseases of the skin in 1958 [5]. BRNS can affect many organs and, since its initial description, it is estimated that there have been fewer than 200 cases published to date [6, 7]. This disease affects both adults and children of both sexes but is rare among black subjects [8]. In most cases, it is not possible to identify a family history, although a dominant pattern of inheritance has been reported [9]. The purpose of this paper is to describe a case of BRNS with cutaneous and colon involvement that evolved from lower gastrointestinal bleeding, the diagnosis of which was confirmed by histopathological study. 2. Case Report A 41-year-old black male was admitted for the investigation of recurrent episodes of lower gastrointestinal bleeding over the previous 10 years

References

[1]  R. Gentry, M. B. Dockerty, and O. T. Clagett, “Vascular malformations and vascular tumours of the gastrointestinal tract,” International Abstracts in Surgery, vol. 88, no. 4, pp. 281–323, 1949.
[2]  M. Camilleri, V. S. Chadwick, and H. J. F. Hodgson, “Vascular anomalies of the gastrointestinal tract,” Hepato-Gastroenterology, vol. 31, no. 3, pp. 149–153, 1984.
[3]  Q. Liu, Y.-P. Chen, and Y.-M. Li, “Blue rubber bleb nevus syndrome: a report of one case associated with recurrent epistaxis,” Chinese Medical Journal, vol. 120, no. 8, pp. 731–733, 2007.
[4]  G. G. Gascoyen, “Case of nevus involving the parotid gland and causing death from suffocation: nevi of the viscera,” Transactions of the Pathological Society of London, vol. 11, article 267, 1860.
[5]  W. B. Bean, “Blue rubber bleb nevi of the skin and gastrointestinal tract,” in Vascular Spiders and Related Lesions of the Skin, pp. 178–185, Charles C. Thomas, Springfield, Ill, USA, 1958.
[6]  M. Moodley and P. Ramdial, “Blue rubber bleb nevus syndrome: case report and review of the literature,” Pediatrics, vol. 92, no. 1, pp. 160–162, 1993.
[7]  Y. Xu, B. Zhou, M. Zhang, and D. Luo, “An unusual case of blue rubber bleb nevus syndrome with unilateral linear distribution,” Indian Journal of Dermatology, Venereology and Leprology, vol. 79, no. 2, pp. 269–270, 2013.
[8]  B. C. ?ksüzo?lu, G. ?ksüzo?lu, U. ?akir, T. Bayir, and M. Esen, “Blue rubber bleb nevus syndrome,” American Journal of Gastroenterology, vol. 91, no. 4, pp. 780–782, 1996.
[9]  V. A. McKusick, “Blue rubber bleb nevus (Bean syndrome),” in Mendelian Inheritance in Man, V. A. McKusick, Ed., pp. 212–213, John Hopkins University Press, Baltimore, Md, USA, 11th edition, 1994.
[10]  M. G. Teixeira, M. V. Perini, C. F. S. Marques, A. Habr-Gama, D. Kiss, and J. J. Gama-Rodrigues, “Blue rubber bleb nevus syndrome: case report,” Revista do Hospital das Clinicas, vol. 58, no. 2, pp. 109–112, 2003.
[11]  Z. Q. Song, Q. Q. Ye, and F. Hao, “Blue rubber bleb nevus syndrome,” Journal of Clinical Dermatology, vol. 33, pp. 213–215, 2004.
[12]  D. Rodrigues, M. L. Bourroul, A. P. Ferrer, H. Monteiro Neto, M. E. Gon?alves, and S. R. Cardoso, “Blue rubber bleb nevus syndrome,” Revista do Hospital das Clinicas de Faculdade de Medicina da Universidade de Sao Paulo, vol. 55, no. 1, pp. 29–34, 2000.
[13]  M. Dòmini, A. Aquino, A. Fakhro et al., “Blue Rubber Bleb Nevus Syndrome and gastrointestinal haemorrhage: which treatment?” European Journal of Pediatric Surgery, vol. 12, no. 2, pp. 129–133, 2002.
[14]  M. D. C. Boente, M. R. Cordisco, M. D. V. Frontini, and R. A. Asial, “Blue rubber bleb nevus (Bean syndrome): evolution of four cases and clinical response to pharmacologic agents,” Pediatric Dermatology, vol. 16, no. 3, pp. 222–227, 1999.
[15]  C. A. McCannel, J. Hoenig, J. Umlas, J. J. Woog, A. N. Newman, and J. B. Bateman, “Orbital lesions in the blue rubber bleb nevus syndrome,” Ophthalmology, vol. 103, no. 6, pp. 933–936, 1996.
[16]  C. Esposito, I. Giurin, A. Farina et al., “Blue rubber bleb nevus: an uncommon cause of intestinal intussusception,” European Journal of Pediatrics, vol. 171, no. 7, pp. 1139–1140, 2012.
[17]  J. L. Beristain-Hernández and A. Rojano-Rodríguez, “Capsule endoscopy images in blue rubber bleb nevus syndrome,” Revista de Gastroenterología de México, vol. 76, no. 4, pp. 362–363, 2011.
[18]  S. Senturk, A. Bilici, T. C. Miroglu, and S. U. Bilek, “Blue rubber bleb nevus syndrome: imaging of small bowel lesions with peroral CT enterography,” Abdominal Imaging, vol. 36, no. 5, pp. 520–523, 2011.
[19]  M. Thomson, K. Venkatesh, K. Elmalik, W. Van Der Veer, and M. Jaacobs, “Double balloon enteroscopy in children: diagnosis, treatment, and safety,” World Journal of Gastroenterology, vol. 16, no. 1, pp. 56–62, 2010.
[20]  J. R. Huo, J. Zhang, X. H. Wang, D. L. Liu, D. L. Ou, and X. W. Liu, “Clinical and endoscopic characteristics in blue rubber bleb nevus syndrome,” Chinese Journal of Digestive Endoscopy, vol. 22, pp. 194–195, 2005.
[21]  P. L. Beck, A. I. Aspinall, V. M. Kilvert, and J. Dort, “Blue rubber bleb nevus syndrome,” Gastrointestinal Endoscopy, vol. 56, no. 4, pp. 598–600, 2002.
[22]  H. Yuksekkaya, O. Ozbek, M. Keser, and H. Toy, “Blue rubber bleb nevus syndrome: successful treatment with sirolimus,” Pediatrics, vol. 129, no. 4, pp. e1080–e1084, 2012.
[23]  O. Enjolras, “Vascular tumors and vascular malformations: are we at the dawn of a better knowledge?” Pediatric Dermatology, vol. 16, no. 3, pp. 238–241, 1999.
[24]  E. K. W. Ng, F. K. Y. Cheung, and P. W. Y. Chiu, “Blue rubber bleb nevus syndrome: treatment of multiple gastrointestinal hemangiomas with argon plasma coagulator,” Digestive Endoscopy, vol. 21, no. 1, pp. 40–42, 2009.
[25]  C. M. Moser and C. Hamsch, “Successful treatment of cutaneous venous malformations in a patient with blue rubber bleb naevus syndrome by Nd:YAG laser,” British Journal of Dermatology, vol. 166, no. 5, pp. 1143–1145, 2012.
[26]  H. Apak, T. Celkan, A. ?zkan et al., “Blue rubber bleb nevus syndrome associated with consumption coagulopathy: treatment with interferon,” Dermatology, vol. 208, no. 4, pp. 345–348, 2004.
[27]  K. K. Choi, J. Y. Kim, M. J. Kim et al., “Radical resection of intestinal blue rubber bleb nevus syndrome,” Journal of the Korean Surgical Society, vol. 83, no. 5, pp. 316–320, 2012.

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