{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,18]],"date-time":"2026-08-18T02:28:26Z","timestamp":1787020106939,"version":"build-2736575974"},"reference-count":13,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2021,9,6]],"date-time":"2021-09-06T00:00:00Z","timestamp":1630886400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2021,9,6]],"date-time":"2021-09-06T00:00:00Z","timestamp":1630886400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"name":"the program of Overseas Students Innovation Project of Anhui Province","award":["No.2019LCX002"],"award-info":[{"award-number":["No.2019LCX002"]}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["BMC Gastroenterol"],"published-print":{"date-parts":[[2021,12]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Hereditary hemorrhagic telangiectasia is an autosomal dominant hereditary hemorrhagic disease. Its main feature is an abnormal structure of the blood vessel wall. Cirrhosis of the liver is a common chronic progressive disease with one or more causes in which diffuse liver damage occurs after long-term or repeated injury. Liver cirrhosis can cause dilation of gastrointestinal capillaries. Many patients with hereditary hemorrhagic telangiectasia accompanied by gastrointestinal vascular malformations and liver cirrhosis may be diagnosed only with liver cirrhosis if the clinician does not pay attention to physical examination findings and family history. Moreover, general treatment measures, such as blood transfusion, iron supplementation, and application of hemostatic drugs, are less effective for bleeding in patients with hereditary hemorrhagic telangiectasia than in those with liver cirrhosis alone.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case presentation<\/jats:title>\n                    <jats:p>Here, we report the rare case of a 75-year-old Chinese man who was admitted to the hospital with repeated melena and epistaxis. He was diagnosed with unexplained liver cirrhosis, which was later confirmed as hereditary hemorrhagic telangiectasia. Subsequently, we implemented the treatment intervention of oral thalidomide combined with gastrointestinal argon plasma coagulation. A follow-up of more than 8\u00a0months showed that the treatment effect was excellent.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>If patients with liver cirrhosis and gastrointestinal vascular malformations also have a family history of epistaxis, special attention should be paid to targeted physical examination results, and the possibility of hereditary hemorrhagic telangiectasia should be considered. Moreover, for patients with hereditary hemorrhagic telangiectasia and both gastrointestinal bleeding caused by gastrointestinal capillaries and repeated epistaxis, when other general treatment measures are ineffective, thalidomide combined with gastrointestinal argon plasma coagulation may be an effective intervention.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1186\/s12876-021-01913-3","type":"journal-article","created":{"date-parts":[[2021,9,6]],"date-time":"2021-09-06T08:03:19Z","timestamp":1630915399000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Hereditary hemorrhagic telangiectasia with liver cirrhosis: a case report"],"prefix":"10.1186","volume":"21","author":[{"given":"Linxia","family":"Xu","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Feng","family":"Xu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Qizhi","family":"Wang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3739-6622","authenticated-orcid":false,"given":"Xiquan","family":"Ke","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2021,9,6]]},"reference":[{"issue":"2","key":"1913_CR1","doi-asserted-by":"publisher","first-page":"73","DOI":"10.1136\/jmg.2009.069013","volume":"48","author":"ME Faughnan","year":"2011","unstructured":"Faughnan ME, Palda VA, Garcia-Tsao G, Geisthoff UW, McDonald J, Proctor DD, et al. International guidelines for the diagnosis and management of hereditary haemorrhagic telangiectasia. J Med Genet. 2011;48(2):73\u201387.","journal-title":"J Med Genet"},{"issue":"1","key":"1913_CR2","doi-asserted-by":"publisher","first-page":"237","DOI":"10.1016\/j.otc.2017.09.017","volume":"51","author":"T K\u00fchnel","year":"2018","unstructured":"K\u00fchnel T, Wirsching K, Wohlgemuth W, Chavan A, Evert K, Vielsmeier V. Hereditary hemorrhagic telangiectasia. Otolaryngol Clin North Am. 2018;51(1):237\u201354.","journal-title":"Otolaryngol Clin North Am"},{"issue":"6","key":"1913_CR3","first-page":"818","volume":"90","author":"H Sadick","year":"2005","unstructured":"Sadick H, Riedel F, Naim R, Goessler U, H\u00f6rmann K, Hafner M, et al. Patients with hereditary hemorrhagic telangiectasia have increased plasma levels of vascular endothelial growth factor and transforming growth factor-beta1 as well as high ALK1 tissue expression. Haematologica. 2005;90(6):818\u201328.","journal-title":"Haematologica"},{"issue":"4","key":"1913_CR4","doi-asserted-by":"publisher","first-page":"1070","DOI":"10.1111\/j.1572-0241.1999.01017.x","volume":"94","author":"F Junquera","year":"1999","unstructured":"Junquera F, Saperas E, de Torres I, Vidal MT, Malagelada JR. Increased expression of angiogenic factors in human colonic angiodysplasia. Am J Gastroenterol. 1999;94(4):1070\u20136.","journal-title":"Am J Gastroenterol"},{"issue":"14","key":"1913_CR5","doi-asserted-by":"publisher","first-page":"918","DOI":"10.1056\/NEJM199510053331407","volume":"333","author":"AE Guttmacher","year":"1995","unstructured":"Guttmacher AE, Marchuk DA, White RI Jr. Hereditary hemorrhagic telangiectasia. 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Arch Surg. 1976;111(4):381\u20139.","journal-title":"Arch Surg"},{"issue":"1","key":"1913_CR8","doi-asserted-by":"publisher","first-page":"58","DOI":"10.1111\/j.1572-0241.2006.00370.x","volume":"101","author":"V Kwan","year":"2006","unstructured":"Kwan V, Bourke MJ, Williams SJ, Gillespie PE, Murray MA, Kaffes AJ, et al. Argon plasma coagulation in the management of symptomatic gastrointestinal vascular lesions: experience in 100 consecutive patients with long-term follow-up. Am J Gastroenterol. 2006;101(1):58\u201363.","journal-title":"Am J Gastroenterol"},{"issue":"10","key":"1913_CR9","doi-asserted-by":"publisher","first-page":"1036","DOI":"10.1055\/s-2006-944829","volume":"38","author":"J Bauditz","year":"2006","unstructured":"Bauditz J, Lochs H, Voderholzer W. Macroscopic appearance of intestinal angiodysplasias under antiangiogenic treatment with thalidomide. Endoscopy. 2006;38(10):1036\u20139.","journal-title":"Endoscopy"},{"key":"1913_CR10","doi-asserted-by":"crossref","unstructured":"Ge ZZ, Chen HM, Gao YJ, Liu WZ, Xu CH, Tan HH, et al. Efficacy of thalidomide for refractory gastrointestinal bleeding from vascular malformation. Gastroenterology. 2011; 141(5):1629\u201337","DOI":"10.1053\/j.gastro.2011.07.018"},{"issue":"4","key":"1913_CR11","doi-asserted-by":"publisher","first-page":"420","DOI":"10.1038\/nm.2131","volume":"16","author":"F Lebrin","year":"2010","unstructured":"Lebrin F, Srun S, Raymond K, Martin S, van den Brink S, Freitas C, et al. Thalidomide stimulates vessel maturation and reduces epistaxis in individuals with hereditary hemorrhagic telangiectasia. Nat Med. 2010;16(4):420\u20138.","journal-title":"Nat Med"},{"issue":"7256","key":"1913_CR12","doi-asserted-by":"publisher","first-page":"599","DOI":"10.1016\/S0140-6736(62)90462-2","volume":"2","author":"JW Millen","year":"1962","unstructured":"Millen JW. Thalidomide and limb deformities. Lancet (London, England). 1962;2(7256):599\u2013600.","journal-title":"Lancet (London, England)"},{"issue":"1","key":"1913_CR13","doi-asserted-by":"publisher","first-page":"28","DOI":"10.1186\/s13023-018-0982-4","volume":"14","author":"E Buscarini","year":"2019","unstructured":"Buscarini E, Botella LM, Geisthoff U, Kjeldsen AD, Mager HJ, Pagella F, et al. Safety of thalidomide and bevacizumab in patients with hereditary hemorrhagic telangiectasia. Orphanet J Rare Dis. 2019;14(1):28.","journal-title":"Orphanet J Rare Dis"}],"container-title":["BMC Gastroenterology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12876-021-01913-3.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s12876-021-01913-3\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12876-021-01913-3.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,9,6]],"date-time":"2021-09-06T08:05:13Z","timestamp":1630915513000},"score":1,"resource":{"primary":{"URL":"https:\/\/bmcgastroenterol.biomedcentral.com\/articles\/10.1186\/s12876-021-01913-3"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2021,9,6]]},"references-count":13,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2021,12]]}},"alternative-id":["1913"],"URL":"https:\/\/doi.org\/10.1186\/s12876-021-01913-3","relation":{},"ISSN":["1471-230X"],"issn-type":[{"value":"1471-230X","type":"electronic"}],"subject":[],"published":{"date-parts":[[2021,9,6]]},"assertion":[{"value":"20 April 2021","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"17 August 2021","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"6 September 2021","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"Not applicable.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"During his lifetime, the patient consented to the use of his history and all related images and information for scientific purposes. After the patient\u2019s death, the patient\u2019s next of kin gave written consent for the publication of the case.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare that they have no competing interests.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"343"}}