{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,10]],"date-time":"2026-09-10T15:46:57Z","timestamp":1789055217032,"version":"build-2803163510"},"reference-count":11,"publisher":"MDPI AG","issue":"1","license":[{"start":{"date-parts":[[2022,12,20]],"date-time":"2022-12-20T00:00:00Z","timestamp":1671494400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Children"],"abstract":"<jats:p>Refractory anemia is not uncommon in pediatric patients, and anemia caused by gastrointestinal tract bleeding should always be kept in mind. Aside from infection or intestinal malrotation related bleeding, vascular malformation should also be considered. Blue rubber bleb nevus syndrome (BRBNS) is a rare vascular disorder consisting of multiple venous malformations. Lack of experience in pediatric BRBNS might lead to delayed diagnosis or misdiagnosis. Herein, we report a case of an eleven-year-old boy with recurrent pallor appearance and weakness diagnosed with BRBNS. After a thorough examination, he was treated with endoscopic polypectomy, and further iron supplements and folic acid. He is now under regular follow-up at our outpatient department. No complication is noted for six months. BRBNS is a rare venous malformation syndrome that mostly involves skin and the gastrointestinal tract. Multidisciplinary approach should be arranged for diagnosis and management. Up to date, no consensus for BRBNS treatment has been reached. Management usually depends on clinical symptoms and severity of damage of involved organs. The options of treatment include conservative, medical, endoscopic, and surgical management.<\/jats:p>","DOI":"10.3390\/children10010003","type":"journal-article","created":{"date-parts":[[2022,12,21]],"date-time":"2022-12-21T02:31:33Z","timestamp":1671589893000},"page":"3","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Blue Rubber Bleb Nevus Syndrome (BRBNS): A Rare Cause of Refractory Anemia in Children"],"prefix":"10.3390","volume":"10","author":[{"given":"Liang-Chun","family":"Chen","sequence":"first","affiliation":[{"name":"Department of Pediatrics, Taipei Hospital, Ministry of Health and Welfare, New Taipei City 242, Taiwan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chun-Yan","family":"Yeung","sequence":"additional","affiliation":[{"name":"Department of Medicine, MacKay Medical College, New Taipei City 252, Taiwan"},{"name":"Department of Pediatric Gastroenterology, Hepatology and Nutrition, Hsinchu Municipal MacKay Children\u2019s Hospital, No. 690, Sec. 2, Guangfu Rd., East Dist., Hsinchu City 300044, Taiwan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chen-Wang","family":"Chang","sequence":"additional","affiliation":[{"name":"Department of Hepatology and Gastroenterology, MacKay Memorial Hospital, Taipei City 104, Taiwan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hung-Chang","family":"Lee","sequence":"additional","affiliation":[{"name":"Department of Pediatric Gastroenterology, Hepatology and Nutrition, MacKay Children\u2019s Hospital, Taipei City 104, Taiwan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9740-800X","authenticated-orcid":false,"given":"Wai-Tao","family":"Chan","sequence":"additional","affiliation":[{"name":"Department of Pediatric Gastroenterology, Hepatology and Nutrition, MacKay Children\u2019s Hospital, Taipei City 104, Taiwan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Chuen-Bin","family":"Jiang","sequence":"additional","affiliation":[{"name":"Department of Pediatric Gastroenterology, Hepatology and Nutrition, MacKay Children\u2019s Hospital, Taipei City 104, Taiwan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Szu-Wen","family":"Chang","sequence":"additional","affiliation":[{"name":"Department of Pediatric Gastroenterology, Hepatology and Nutrition, Mackay Memorial Tamshui Branch Hospital, New Taipei City 251, Taiwan"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2022,12,20]]},"reference":[{"key":"ref_1","doi-asserted-by":"crossref","first-page":"517","DOI":"10.1002\/jcu.22000","article-title":"Association of blue rubber bleb nevus syndrome and placenta previa: Report of a case","volume":"41","author":"Terata","year":"2013","journal-title":"J. Clin. Ultrasound."},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"2960","DOI":"10.21037\/tp-21-238","article-title":"Blue rubber bleb nevus syndrome: A single-center case series in 12 years","volume":"10","author":"Xia","year":"2021","journal-title":"Transl. Pediatr."},{"key":"ref_3","doi-asserted-by":"crossref","first-page":"e3","DOI":"10.1053\/j.gastro.2008.05.008","article-title":"Electronic clinical challenges and images in GI. Blue rubber bleb nevus syndrome","volume":"134","author":"Yen","year":"2008","journal-title":"Gastroenterology"},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"e4","DOI":"10.1053\/j.gastro.2017.05.025","article-title":"A rare cause of chronic anemia and recurrent bowel obstruction","volume":"154","author":"Chen","year":"2018","journal-title":"Gastroenterology"},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"600","DOI":"10.1007\/BF02012746","article-title":"Gastrointestinal bleeding and paraparesis in blue rubber bleb nevus syndrome","volume":"24","author":"Wong","year":"1994","journal-title":"Pediatr. Radiol."},{"key":"ref_6","first-page":"683684","article-title":"Blue rubber bleb nevus syndrome as a cause of lower digestive bleeding","volume":"2014","author":"Martinez","year":"2014","journal-title":"Case Rep. Surg."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"207","DOI":"10.1016\/j.jid.2016.07.034","article-title":"Blue rubber bleb nevus (BRBN) syndrome is caused by somatic TEK (TIE2) mutations","volume":"137","author":"Soblet","year":"2017","journal-title":"J. Investig. Dermatol."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"1583","DOI":"10.1093\/hmg\/3.9.1583","article-title":"Assignment of a locus for dominantly inherited venous malformations to chromosome 9p","volume":"3","author":"Boon","year":"1994","journal-title":"Hum. Mol. Genet."},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"197","DOI":"10.1136\/jmg.32.3.197","article-title":"A gene for familial venous malformations maps to chromosome 9p in a second large kindred","volume":"32","author":"Gallione","year":"1995","journal-title":"J. Med. Genet."},{"key":"ref_10","doi-asserted-by":"crossref","first-page":"152","DOI":"10.1111\/jpc.14345","article-title":"Sirolimus in blue rubber bleb naevus syndrome: A systematic review","volume":"55","author":"Wong","year":"2019","journal-title":"J. Paediatr. Child Health"},{"key":"ref_11","doi-asserted-by":"crossref","first-page":"e1080","DOI":"10.1542\/peds.2010-3611","article-title":"Blue rubber bleb nevus syndrome: Successful treatment with sirolimus","volume":"129","author":"Yuksekkaya","year":"2012","journal-title":"Pediatrics"}],"container-title":["Children"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2227-9067\/10\/1\/3\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,11]],"date-time":"2025-10-11T01:44:43Z","timestamp":1760147083000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2227-9067\/10\/1\/3"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2022,12,20]]},"references-count":11,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2023,1]]}},"alternative-id":["children10010003"],"URL":"https:\/\/doi.org\/10.3390\/children10010003","relation":{},"ISSN":["2227-9067"],"issn-type":[{"value":"2227-9067","type":"electronic"}],"subject":[],"published":{"date-parts":[[2022,12,20]]}}}