Abstract
Blue rubber bleb nevus syndrome (BRBNS) is a rare vascular disorder consisting of multifocal venous malformations. Delayed diagnosis or misdiagnosis frequently occurs in patients without typical cutaneous lesions or gastrointestinal bleeding symptoms. This article reports a 10-year case of delayed diagnosis of BRBNS detected by capsule endoscopy. A 15-year-old girl presented with refractory iron-deficiency anemia (IDA) for 10 years, without any hemorrhagic signs or noticeable cutaneous lesions, which led to her obvious physical growth retardation. Capsule endoscopic examination revealed dozens of vascular blebs distributed from the jejunum to the ileum and a site of active bleeding. Hence, she was diagnosed with BRBNS. Laparotomy was performed with resection of the small bowel lesions, and iron supplementation was prescribed for 3 months. Postoperatively, the patient had an uncomplicated course. On follow-up after 3 years, IDA in this patient was cured and she did not require further blood transfusion and showed excellent vigor. A high index of suspicion for BRBNS and adequate endoscopy examination will help to identify the origin of refractory IDA in older children, particularly in patients with vascular lesions of the skin.
References
Sep 23, 2003·AJR. American Journal of Roentgenology·Ara KassarjianPatricia E Burrows
Dec 5, 2009·Acta Paediatrica·M AgneseA Staiano
Dec 9, 2010·Rare Tumors·Carolin MoglerKai Breuhahn
Mar 7, 2012·Pediatrics·Hasan YuksekkayaHatice Toy
Nov 21, 2012·Journal of the Korean Surgical Society·Kang Kook ChoiJin Seok Heo
Mar 23, 2013·Clinics and Research in Hepatology and Gastroenterology·Rtika R AbrahamMohit Girotra
Nov 14, 2013·Experimental and Therapeutic Medicine·Weiwei GuoZheng Liu
Dec 11, 2014·World Journal of Gastroenterology : WJG·Xue-Li JinXiao-Ying Zhao
Apr 18, 2015·Indian Journal of Pathology & Microbiology·Ankur KumarKanchan Jain
Feb 24, 2016·Annals of Medicine and Surgery·Zhi LiXiaoyi Sun
Feb 26, 2016·Journal of Community Hospital Internal Medicine Perspectives·Aditya GoudStephen Selinger
Apr 28, 2016·Pediatrics International : Official Journal of the Japan Pediatric Society·Takehiko DoiYuji Mizuno
Aug 16, 2016·The Journal of Investigative Dermatology·Julie SobletMiikka Vikkula
Dec 22, 2016·BMJ Case Reports·Martin Bell LybeckerNiels Clausen
Sep 1, 2017·International Journal of Surgery Case Reports·Carlos Augusto Metidieri MenegozzoEdivaldo Massazo Utiyama
Citations
Sep 18, 2020·Child's Nervous System : ChNS : Official Journal of the International Society for Pediatric Neurosurgery·Martino RuggieriConcezio Di Rocco
Dec 19, 2019·BMJ Case Reports·Mamoon Ur RashidAbu Hurairah
Jul 16, 2020·Medicine·Zeming HuJianhong Zhang
Jan 1, 2019·Case Reports in Gastrointestinal Medicine·Hideaki NakajimaYutaka Yamada
Mar 23, 2021·Dermatologic Therapy·Andrea DiociaiutiMay El Hachem
Jun 15, 2021·ANZ Journal of Surgery·Xia MengZiqiang Wang
Oct 2, 2021·Radiographics : a Review Publication of the Radiological Society of North America, Inc·Lisa QiuJonathan R Dillman