{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,25]],"date-time":"2026-10-25T03:48:45Z","timestamp":1792900125804,"version":"4.5.0"},"reference-count":26,"publisher":"Cambridge University Press (CUP)","issue":"10","license":[{"start":{"date-parts":[[2018,9,7]],"date-time":"2018-09-07T00:00:00Z","timestamp":1536278400000},"content-version":"unspecified","delay-in-days":0,"URL":"https:\/\/www.cambridge.org\/core\/terms"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["J. Laryngol. Otol."],"published-print":{"date-parts":[[2018,10]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec id=\"S0022215118001536_sec_a1\"><jats:title>Background<\/jats:title><jats:p>Hereditary haemorrhagic telangiectasia is an autosomal dominant condition, characterised by mucocutaneous telangiectasia, aneurysm and arteriovenous malformations. Thalidomide has been used as a therapeutic strategy for refractory epistaxis in hereditary haemorrhagic telangiectasia patients. This review set out to examine the evidence for using thalidomide in the management of refractory epistaxis in hereditary haemorrhagic telangiectasia patients.<\/jats:p><\/jats:sec><jats:sec id=\"S0022215118001536_sec_a2\" sec-type=\"methods\"><jats:title>Methods<\/jats:title><jats:p>A systematic search of the available literature was performed using Medline, Embase, Cochrane Library and NHS Evidence databases, from inception to December 2017. The search terms used included: hereditary haemorrhagic telangiectasia (HHT), Osler-Weber-Rendu syndrome, epistaxis, haemorrhage and thalidomide.<\/jats:p><\/jats:sec><jats:sec id=\"S0022215118001536_sec_a3\" sec-type=\"results\"><jats:title>Results<\/jats:title><jats:p>All studies using thalidomide therapy showed a reduction in the frequency and duration of epistaxis, as early as four weeks post-therapy. In addition, thalidomide therapy was shown to increase median haemoglobin levels and reduce blood transfusion dependence.<\/jats:p><\/jats:sec><jats:sec id=\"S0022215118001536_sec_a4\" sec-type=\"conclusion\"><jats:title>Conclusion<\/jats:title><jats:p>Current available evidence suggests that low-dose thalidomide is effective in transiently reducing epistaxis frequency and duration. Further studies are required to establish a treatment regimen to prevent side effects.<\/jats:p><\/jats:sec>","DOI":"10.1017\/s0022215118001536","type":"journal-article","created":{"date-parts":[[2018,9,7]],"date-time":"2018-09-07T10:21:35Z","timestamp":1536315695000},"page":"866-871","source":"Crossref","is-referenced-by-count":25,"title":["The use of thalidomide therapy for refractory epistaxis in hereditary haemorrhagic telangiectasia: systematic review"],"prefix":"10.1017","volume":"132","author":[{"given":"L","family":"Harrison","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A","family":"Kundra","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"P","family":"Jervis","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"56","published-online":{"date-parts":[[2018,9,7]]},"reference":[{"key":"S0022215118001536_ref25","first-page":"40","article-title":"Natural history and control of epistaxis in a group of German patients with Rendu-Osler-Weber disease","volume":"43","author":"Folz","year":"2005","journal-title":"Rhinology"},{"key":"S0022215118001536_ref15","doi-asserted-by":"publisher","DOI":"10.1016\/S1359-6446(04)03307-0"},{"key":"S0022215118001536_ref5","doi-asserted-by":"publisher","DOI":"10.1016\/S0140-6736(04)16308-3"},{"key":"S0022215118001536_ref21","doi-asserted-by":"crossref","first-page":"1031","DOI":"10.1160\/TH07-01-0064","article-title":"Elevated factor VIII in hereditary haemorrhagic telangiectasia (HHT): association with venous thromboembolism","volume":"98","author":"Shovlin","year":"2007","journal-title":"Thromb Haemost"},{"key":"S0022215118001536_ref11","first-page":"21","article-title":"Using thalidomide in a patient with epithelioid leiomyosarcoma and Osler-Weber-Rendu disease","volume":"16","author":"Kurstin","year":"2002","journal-title":"Oncology"},{"key":"S0022215118001536_ref2","doi-asserted-by":"publisher","DOI":"10.1002\/alr.21374"},{"key":"S0022215118001536_ref1","doi-asserted-by":"publisher","DOI":"10.1007\/s11239-012-0840-5"},{"key":"S0022215118001536_ref13","doi-asserted-by":"publisher","DOI":"10.3389\/fgene.2015.00037"},{"key":"S0022215118001536_ref3","doi-asserted-by":"publisher","DOI":"10.1007\/s11136-004-7865-y"},{"key":"S0022215118001536_ref23","unstructured":"National Institute for Health and Care Excellence. Bortezomib and thalidomide for the first-line treatment of multiple myeloma. In: https:\/\/www.nice.org.uk\/guidance\/ta228 [20 August 2018]"},{"key":"S0022215118001536_ref22","unstructured":"International Myeloma Foundation. Understanding Thalidomide Therapy. In: https:\/\/www.myeloma.org\/sites\/default\/files\/images\/publications\/UnderstandingPDF\/u-thalidomide.pdf [20 August 2018]"},{"key":"S0022215118001536_ref6","doi-asserted-by":"publisher","DOI":"10.1038\/nm.2131"},{"key":"S0022215118001536_ref7","doi-asserted-by":"publisher","DOI":"10.1016\/S2352-3026(15)00195-7"},{"key":"S0022215118001536_ref8","doi-asserted-by":"crossref","first-page":"3050","DOI":"10.4103\/0366-6999.169068","article-title":"Thalidomide effects in patients with hereditary haemorrhagic telangiectasia during therapeutic treatment in Fli-EGFP transgenic zebrafish model","volume":"128","author":"Peng","year":"2015","journal-title":"Chin Med J (Engl)"},{"key":"S0022215118001536_ref12","doi-asserted-by":"publisher","DOI":"10.1177\/0194599817700573"},{"key":"S0022215118001536_ref17","doi-asserted-by":"publisher","DOI":"10.1007\/s12185-008-0220-2"},{"key":"S0022215118001536_ref24","doi-asserted-by":"publisher","DOI":"10.1093\/qjmed\/hci148"},{"key":"S0022215118001536_ref26","doi-asserted-by":"publisher","DOI":"10.1288\/00005537-199109000-00008"},{"key":"S0022215118001536_ref4","doi-asserted-by":"publisher","DOI":"10.3389\/fgene.2015.00001"},{"key":"S0022215118001536_ref19","doi-asserted-by":"publisher","DOI":"10.1016\/j.ijpharm.2016.07.002"},{"key":"S0022215118001536_ref10","doi-asserted-by":"publisher","DOI":"10.1097\/MBC.0b013e32834a040c"},{"key":"S0022215118001536_ref9","doi-asserted-by":"publisher","DOI":"10.4193\/Rhin14.289"},{"key":"S0022215118001536_ref14","doi-asserted-by":"publisher","DOI":"10.1136\/jmg.40.8.585"},{"key":"S0022215118001536_ref20","doi-asserted-by":"publisher","DOI":"10.1136\/thoraxjnl-2011-201076"},{"key":"S0022215118001536_ref16","doi-asserted-by":"publisher","DOI":"10.1111\/j.1529-8027.2008.00193.x"},{"key":"S0022215118001536_ref18","doi-asserted-by":"publisher","DOI":"10.1111\/imj.12850"}],"container-title":["The Journal of Laryngology &amp; Otology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.cambridge.org\/core\/services\/aop-cambridge-core\/content\/view\/S0022215118001536","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2019,4,13]],"date-time":"2019-04-13T19:50:22Z","timestamp":1555185022000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.cambridge.org\/core\/product\/identifier\/S0022215118001536\/type\/journal_article"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2018,9,7]]},"references-count":26,"journal-issue":{"issue":"10","published-print":{"date-parts":[[2018,10]]}},"alternative-id":["S0022215118001536"],"URL":"https:\/\/doi.org\/10.1017\/s0022215118001536","relation":{},"ISSN":["0022-2151","1748-5460"],"issn-type":[{"value":"0022-2151","type":"print"},{"value":"1748-5460","type":"electronic"}],"subject":[],"published":{"date-parts":[[2018,9,7]]}}}