{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,2,21]],"date-time":"2025-02-21T08:15:39Z","timestamp":1740125739435,"version":"3.37.3"},"reference-count":45,"publisher":"Wiley","issue":"3","license":[{"start":{"date-parts":[[2015,4,9]],"date-time":"2015-04-09T00:00:00Z","timestamp":1428537600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100007658","name":"Baxter Healthcare Corporation","doi-asserted-by":"publisher","id":[{"id":"10.13039\/100007658","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["Haemophilia"],"published-print":{"date-parts":[[2015,5]]},"abstract":"<jats:sec><jats:title>\n<jats:bold>Objective:<\/jats:bold>\n<\/jats:title><jats:p>A cost analysis model was developed to compare annual cost of prophylaxis with activated prothrombin complex concentrate (<jats:styled-content style=\"fixed-case\">aPCC<\/jats:styled-content>) vs. on\u2010demand therapy with activated recombinant factor <jats:styled-content style=\"fixed-case\">VII<\/jats:styled-content> (<jats:styled-content style=\"fixed-case\">rFVII<\/jats:styled-content>a) in severe haemophilia A patients with inhibitors for the Spanish National Health System (<jats:styled-content style=\"fixed-case\">NHS<\/jats:styled-content>).<\/jats:p><\/jats:sec><jats:sec><jats:title>\n<jats:bold>Methods:<\/jats:bold>\n<\/jats:title><jats:p>Model inputs were drug cost for prophylaxis (<jats:styled-content style=\"fixed-case\">aPCC<\/jats:styled-content>) and for on\u2010demand treatment (<jats:styled-content style=\"fixed-case\">rFVII<\/jats:styled-content>a or <jats:styled-content style=\"fixed-case\">aPCC<\/jats:styled-content>); bleeding episodes management (excluding bypassing agent cost); surgical costs and disease management (excluding bleeding episodes). Annual bleeding episodes treated on\u2010demand was assumed to be 25, whereas breakthrough bleeds on prophylaxis was 8. Dose for prophylaxis was 75.72\u00a0U\u00a0kg<jats:sup>\u22121<\/jats:sup>, three\u00a0times per week. The total on\u2010demand dose\/bleeding episode was 679.66\u00a0\u03bcg\u00a0kg<jats:sup>\u22121<\/jats:sup> (<jats:styled-content style=\"fixed-case\">rFVII<\/jats:styled-content>a) and 235.28\u00a0U\u00a0kg<jats:sup>\u22121<\/jats:sup> (<jats:styled-content style=\"fixed-case\">aPCC<\/jats:styled-content>). The average bleeding cost (\u20ac2998) considered different bleeding sites (62.5% joints, 28.6% muscles and soft tissues, 3.6% mucocutaneous tissues and 5.4% other areas). A 7.5% deduction was applied to ex\u2010factory drug prices. Unitary costs (\u20ac2013) derived from local databases. Sensitivity analyses (<jats:styled-content style=\"fixed-case\">SA<\/jats:styled-content>) were performed.<\/jats:p><\/jats:sec><jats:sec><jats:title>\n<jats:bold>Results:<\/jats:bold>\n<\/jats:title><jats:p>Annual cost of <jats:styled-content style=\"fixed-case\">aPCC<\/jats:styled-content> prophylaxis (\u20ac524\u00a0358) was 16% lower than on\u2010demand treatment with <jats:styled-content style=\"fixed-case\">rFVII<\/jats:styled-content>a (\u20ac627\u00a0876). Yearly drug costs were \u20ac497\u00a0017 for <jats:styled-content style=\"fixed-case\">aPCC<\/jats:styled-content> (\u20ac73\u00a0166 for on\u2010demand treatment and \u20ac423\u00a0850 for prophylaxis), and \u20ac548\u00a0870 for <jats:styled-content style=\"fixed-case\">rFVII<\/jats:styled-content>a. Disease management cost (\u20ac2645 per year) and surgical procedures (\u20ac708 per year) were common for both strategies. In the <jats:styled-content style=\"fixed-case\">SA<\/jats:styled-content> prophylactic treatment led to savings\u00a0between \u20ac26\u00a0225 and \u20ac\u20101\u00a0008\u00a0960.<\/jats:p><\/jats:sec><jats:sec><jats:title><jats:bold>Conclusion<\/jats:bold>:<\/jats:title><jats:p>Prophylaxis with <jats:styled-content style=\"fixed-case\">aPCC<\/jats:styled-content> reduces number of bleeding episodes in severe haemophilia A patients with inhibitors. <jats:styled-content style=\"fixed-case\">aPCC<\/jats:styled-content> prophylaxis resulted in savings in excess of \u20ac100\u00a0000 per\u2010patient per year, being 16% less costly than on\u2010demand treatment with <jats:styled-content style=\"fixed-case\">rFVII<\/jats:styled-content>a, for the Spanish NHS.<\/jats:p><\/jats:sec>","DOI":"10.1111\/hae.12681","type":"journal-article","created":{"date-parts":[[2015,4,9]],"date-time":"2015-04-09T08:00:59Z","timestamp":1428566459000},"page":"320-329","source":"Crossref","is-referenced-by-count":12,"title":["Cost analysis of prophylaxis with activated prothrombin complex concentrate vs. on\u2010demand therapy with activated factor <scp>VII<\/scp> in severe haemophilia A patients with inhibitors, in Spain"],"prefix":"10.1111","volume":"21","author":[{"given":"R.","family":"Villarrubia","sequence":"first","affiliation":[{"name":"Pharmacoeconomics &amp; Outcomes Research Iberia Madrid Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"I.","family":"Oyag\u00fcez","sequence":"additional","affiliation":[{"name":"Pharmacoeconomics &amp; Outcomes Research Iberia Madrid Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"M. T.","family":"\u00c1lvarez\u2010Rom\u00e1n","sequence":"additional","affiliation":[{"name":"La Paz University Hospital Madrid Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"M. E.","family":"Mingot\u2010Castellano","sequence":"additional","affiliation":[{"name":"Malaga Regional University Hospital M\u00e1laga Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"R.","family":"Parra","sequence":"additional","affiliation":[{"name":"Vall d'Hebron University Hospital Barcelona Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"M. A.","family":"Casado","sequence":"additional","affiliation":[{"name":"Pharmacoeconomics &amp; Outcomes Research Iberia Madrid Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"311","published-online":{"date-parts":[[2015,4,9]]},"reference":[{"key":"e_1_2_8_2_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2012.02909.x"},{"key":"e_1_2_8_3_1","doi-asserted-by":"publisher","DOI":"10.1586\/ehm.12.27"},{"key":"e_1_2_8_4_1","doi-asserted-by":"publisher","DOI":"10.1155\/2012\/201271"},{"key":"e_1_2_8_5_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2516.2002.00591.x"},{"key":"e_1_2_8_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2009.02127.x"},{"key":"e_1_2_8_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2009.02001.x"},{"key":"e_1_2_8_8_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2009.02101.x"},{"key":"e_1_2_8_9_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1538-7836.2011.04214.x"},{"key":"e_1_2_8_10_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa067659"},{"key":"e_1_2_8_11_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12178"},{"key":"e_1_2_8_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/0140-6736(92)90874-3"},{"key":"e_1_2_8_13_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2516.2003.00780.x"},{"key":"e_1_2_8_14_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2516.1998.440558.x"},{"key":"e_1_2_8_15_1","doi-asserted-by":"publisher","DOI":"10.1046\/j.1365-2516.1999.00034.x"},{"key":"e_1_2_8_16_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2141.2006.06087.x"},{"key":"e_1_2_8_17_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2006.01359.x"},{"key":"e_1_2_8_18_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2011-08-369132"},{"key":"e_1_2_8_19_1","doi-asserted-by":"publisher","DOI":"10.1053\/j.seminhematol.2006.03.004"},{"key":"e_1_2_8_20_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2007.01497.x"},{"key":"e_1_2_8_21_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2012.02790.x"},{"key":"e_1_2_8_22_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2003-03-0941"},{"key":"e_1_2_8_23_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood-2006-04-017988"},{"key":"e_1_2_8_24_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2006.01284.x"},{"key":"e_1_2_8_25_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1538-7836.2004.00759.x"},{"key":"e_1_2_8_26_1","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa1104435"},{"key":"e_1_2_8_27_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12246"},{"key":"e_1_2_8_28_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12390"},{"key":"e_1_2_8_29_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2007.01442.x"},{"key":"e_1_2_8_30_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2009.01980.x"},{"key":"e_1_2_8_31_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2009.02116.x"},{"key":"e_1_2_8_32_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2009.02126.x"},{"key":"e_1_2_8_33_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.thromres.2012.08.305"},{"key":"e_1_2_8_34_1","first-page":"113","article-title":"Evaluation of FEIBA for prophylaxis in patients with inhibitors","volume":"10","author":"Ewenstein B","year":"2004","journal-title":"Haemophilia"},{"key":"e_1_2_8_35_1","article-title":"FEIBA prophylaxis in hemophilia A patient with inhibitors decreases bleeding episodes, improves joint function and enhance quality of life","volume":"12","author":"Cheng S","year":"2006","journal-title":"Haemophilia"},{"key":"e_1_2_8_36_1","article-title":"Anamnesis in patients with hemophilia and inhibitors who receive activated prothrombin complex concentrates for prophylaxis","volume":"5","author":"Ewing NP","year":"2007","journal-title":"J Thromb Haemost"},{"key":"e_1_2_8_37_1","article-title":"Can long\u2010term prophylaxis with APCC improve the bleeding rate and quality of life of frequently bleeding haemophiliacs with inhibitor?","volume":"12","author":"Lambert T","year":"2006","journal-title":"Haemophilia"},{"key":"e_1_2_8_38_1","unstructured":"BotPlus 2.0.Consejo General de Colegios Oficiales de Farmac\u00e9uticos. Available at:https:\/\/botplusweb.portalfarma.com\/botplus.aspx. Accessed October 23 2013."},{"key":"e_1_2_8_39_1","unstructured":"Oblikue \u2010 Database on costs and prices of the Spanish healthcare sector. Available at:http:\/\/www.oblikue.com\/bddcostes\/. Accessed October 23 2013."},{"key":"e_1_2_8_40_1","unstructured":"KonkleBA JosephsonNC NakayaFletcher SM ThompsonAR.Hemophilia A. [updated 2011 Sep 22]. In:PagonRA AdamMP BirdTD DolanCR FongCT SmithRJH StephensKeds. GeneReviews\u00ae [Internet].Seattle WA:University of Washington Seattle 2000 1993\u20132014. Available athttp:\/\/www.ncbi.nlm.nih.gov\/books\/NBK1404\/. Accessed on March 17 2014."},{"key":"e_1_2_8_41_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2007.01455.x"},{"key":"e_1_2_8_42_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2009.02100.x"},{"key":"e_1_2_8_43_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2010.02231.x"},{"key":"e_1_2_8_44_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2008.01915.x"},{"key":"e_1_2_8_45_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1365-2516.2011.02611.x"},{"key":"e_1_2_8_46_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajh.1146"}],"container-title":["Haemophilia"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/api.wiley.com\/onlinelibrary\/tdm\/v1\/articles\/10.1111%2Fhae.12681","content-type":"unspecified","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.12681","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/hae.12681","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.12681","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,8,24]],"date-time":"2023-08-24T12:32:35Z","timestamp":1692880355000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/hae.12681"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2015,4,9]]},"references-count":45,"journal-issue":{"issue":"3","published-print":{"date-parts":[[2015,5]]}},"alternative-id":["10.1111\/hae.12681"],"URL":"https:\/\/doi.org\/10.1111\/hae.12681","archive":["Portico"],"relation":{},"ISSN":["1351-8216","1365-2516"],"issn-type":[{"type":"print","value":"1351-8216"},{"type":"electronic","value":"1365-2516"}],"subject":[],"published":{"date-parts":[[2015,4,9]]}}}