{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,16]],"date-time":"2026-10-16T03:37:36Z","timestamp":1792121856216,"version":"4.4.0"},"reference-count":21,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2026,4,24]],"date-time":"2026-04-24T00:00:00Z","timestamp":1776988800000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,4,24]],"date-time":"2026-04-24T00:00:00Z","timestamp":1776988800000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Haemophilia"],"published-print":{"date-parts":[[2026,7]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Inhibition of tissue factor pathway inhibitor represents an advanced rebalancing strategy for hemophilia, applicable across hemophilia A and B irrespective of inhibitor status. Although concizumab has demonstrated efficacy in clinical trials, real\u2010world data remain very limited.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Objectives<\/jats:title>\n                    <jats:p>To evaluate the real\u2010world effectiveness and safety of concizumab prophylaxis in an unselected national cohort of patients treated in routine clinical practice in France.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>This cross\u2010sectional observational study included all patients receiving concizumab through compassionate\u2010use or Early Access Programs across six French hemophilia centers. Retrospective data were collected from treatment initiation to a predefined cutoff (1, December 2025). Outcomes included bleeding rates, target joint status, surgical management, laboratory monitoring practices, adherence, and adverse events.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Eleven patients were included, ten with hemophilia B with inhibitors and one with hemophilia B without inhibitors. Mean exposure to concizumab was 22.8 months. Compared with the pre\u2010treatment period, concizumab prophylaxis was associated with a marked reduction in bleeding, including complete absence of bleeding in four patients and resolution of target joints in nine of ten patients. Nine surgical procedures, including two major surgeries, were performed under concizumab prophylaxis with concomitant rFVIIa without thrombotic complications. No thrombotic events or injection\u2010site pain were reported. None of the patients discontinued concizumab prophylaxis.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>In this nationwide real\u2010world cohort, concizumab prophylaxis was effective, well tolerated, and manageable in routine practice, including during surgical procedures. These data support its use as a valuable prophylactic option in carefully selected patients with hemophilia B and inhibitors, while underscoring the importance of individualized monitoring and thrombotic risk assessment.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/hae.70290","type":"journal-article","created":{"date-parts":[[2026,4,24]],"date-time":"2026-04-24T11:01:29Z","timestamp":1777028489000},"page":"914-922","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Real\u2010World Effectiveness and Safety of Concizumab Prophylaxis in Hemophilia: Results From a National French Cohort"],"prefix":"10.1111","volume":"32","author":[{"given":"Caroline","family":"Galeotti","sequence":"first","affiliation":[{"name":"Centre De R\u00e9f\u00e9rence de L'h\u00e9mophilie Et Des Maladies H\u00e9morragiques Rares H\u00f4pital Bic\u00eatre, Universit\u00e9 Paris\u2010Saclay  Le KremlinBic\u00eatre France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Antoine","family":"Rauch","sequence":"additional","affiliation":[{"name":"H\u00e9matologie\u2010transfusion, CHU De Lille  Lille France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Helene","family":"Gatti","sequence":"additional","affiliation":[{"name":"Service De P\u00e9diatrie, Hopital Taaone  Pirae Polynesie Francaise"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Stephanie","family":"Desage","sequence":"additional","affiliation":[{"name":"Centre De Reference de L'hemophilie \u2010 Unite d'Hemostase Clinique Hopital Louis Pradel Et UR4609 Hemostase &amp; Thrombose Universite Lyon 1  Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Alexandre","family":"Butelet","sequence":"additional","affiliation":[{"name":"Service H\u00e9matologie Biologique UF H\u00e9mostase clinique, H\u00f4pital Robert, Debr\u00e9  CHU Reims France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Christine","family":"Biron\u2010Andreani","sequence":"additional","affiliation":[{"name":"Centre De Ressources et De Comp\u00e9tences \u2010 Maladies H\u00e9morragiques rares Service D'h\u00e9matologie Biologique, CHU De Montpellier  Montpellier France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sophie","family":"Susen","sequence":"additional","affiliation":[{"name":"H\u00e9matologie\u2010transfusion, CHU De Lille  Lille France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Roseline","family":"d'Oiron","sequence":"additional","affiliation":[{"name":"Centre De R\u00e9f\u00e9rence de L'h\u00e9mophilie Et Des Maladies H\u00e9morragiques Rares H\u00f4pital Bic\u00eatre, Universit\u00e9 Paris\u2010Saclay  Le KremlinBic\u00eatre France"},{"name":"Inserm H\u00e9mostase inflammation Thrombose HITH U1176 Universit\u00e9 Paris\u2010Saclay  Le Kremlin\u2010Bic\u00eatre France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Guillaume","family":"Chanteau","sequence":"additional","affiliation":[{"name":"Hematologie Clinique, Hopital Taaone  PAPEETE Polynesie Francaise"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Valerie","family":"Proulle","sequence":"additional","affiliation":[{"name":"Service H\u00e9matologie Biologique UF H\u00e9mostase clinique, H\u00f4pital Robert, Debr\u00e9  CHU Reims France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yesim","family":"Dargaud","sequence":"additional","affiliation":[{"name":"Centre De Reference de L'hemophilie \u2010 Unite d'Hemostase Clinique Hopital Louis Pradel Et UR4609 Hemostase &amp; Thrombose Universite Lyon 1  Lyon France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,4,24]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13111"},{"key":"e_1_2_10_3_1","doi-asserted-by":"publisher","DOI":"10.1182\/blood.2019000658"},{"key":"e_1_2_10_4_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jtha.2022.12.029"},{"issue":"5","key":"e_1_2_10_5_1","first-page":"1334","article-title":"Emicizumab: The Hemophilia A Game\u2010changer","volume":"109","author":"Alcedo Andrade P. 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