{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,19]],"date-time":"2026-09-19T10:12:53Z","timestamp":1789812773525,"version":"4.0.1"},"reference-count":24,"publisher":"Wiley","issue":"5","license":[{"start":{"date-parts":[[2026,5,31]],"date-time":"2026-05-31T00:00:00Z","timestamp":1780185600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"},{"start":{"date-parts":[[2026,5,31]],"date-time":"2026-05-31T00:00:00Z","timestamp":1780185600000},"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":[[2026,9]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Introduction<\/jats:title>\n                    <jats:p>Patients with von Willebrand disease (VWD) are prone to bleeding, yet they may also experience thrombotic events, whose nature, frequency, and optimal management remain poorly characterised.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Aim<\/jats:title>\n                    <jats:p>To describe the nature and frequency of venous and arterial thrombotic events in VWD patients.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>This multicentre retrospective study analyzed thrombotic events in 1345 patients with constitutional VWD and von Willebrand factor (VWF) activity \u226430\u00a0IU\/dL from the French BERHLINGO database. Venous events included deep vein thrombosis (DVT) and pulmonary embolism (PE); arterial events included angina, myocardial infarction (MI), ischaemic stroke (ICVA), transient ischaemic attack (TIA), and peripheral artery disease (PAD). Risk factors, therapeutic strategies, efficacy, and bleeding complications were also assessed.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>We identified 35 thrombotic events: 30 arterial (12 angina, 6 MI, 7 PAD, 4 ICVA, 1 TIA) in 20 patients, and 5 venous (3 PE\u00b1DVT, 2 DVT) in 4 patients (1.8% prevalence). The mean patient age was 61.8\u00b114 years. Most arterial events (70%) occurred in men; all venous events were provoked in women. Therapeutic adjustments were made for 10 arterial events (28.6%: 4 withholding, 6 low\u2010dose). Of the 35 events, 11 (31.4%) were recurrences (second or subsequent) in the same patient. Overall, 8\/24 patients (33.3%) had multiple events, always at the same site, including twice (18.2%) after therapeutic adjustments. Only trauma\u2010induced bleeding was reported.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Although rare, thrombosis in VWD patients is associated with age and gender. Given the low bleeding risk, therapeutic approaches similar to those in the general population may be considered.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Trial Registration<\/jats:title>\n                    <jats:p>Cardiovascular and Venous Thromboembolism Disease in Patients with Von Willebrand Disease in the French West (TWIGO); ClinicalTrials.gov ID NCT05773638<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/hae.70309","type":"journal-article","created":{"date-parts":[[2026,6,1]],"date-time":"2026-06-01T06:24:21Z","timestamp":1780295061000},"page":"1208-1221","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Venous and Arterial Thrombo\u2010Embolic Events in Patients With von Willebrand Disease From Western France: The TWIGO Study"],"prefix":"10.1111","volume":"32","author":[{"ORCID":"https:\/\/orcid.org\/0009-0000-3066-7816","authenticated-orcid":false,"given":"Agn\u00e8s","family":"Granottier","sequence":"first","affiliation":[{"name":"Haemophilia Treatment Centre CRC\u2010MHC, Haematology University Hospital of Brest  Brest France"},{"name":"Internal Medicine Department University Hospital of Brest  Brest France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kilian","family":"Trin","sequence":"additional","affiliation":[{"name":"INSERM Clinical Investigation Centre 1412 University Hospital of Brest  Brest France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4322-4544","authenticated-orcid":false,"given":"Sara","family":"Robin","sequence":"additional","affiliation":[{"name":"Internal Medicine Department University Hospital of Brest  Brest France"},{"name":"INSERM, UMR 1304\u2010GETBO Universit\u00e9 de Brest Brest France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-4072-839X","authenticated-orcid":false,"given":"Marc","family":"Trossaert","sequence":"additional","affiliation":[{"name":"Haemophilia Treatment Centre CRC\u2010MHC University Hospital of Nantes  Nantes France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laurent","family":"Ardillon","sequence":"additional","affiliation":[{"name":"Haemophilia Treatment Centre CRC\u2010MHC University Hospital of Tours  Tours France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Damien","family":"Tabaux","sequence":"additional","affiliation":[{"name":"Haemophilia Treatment Centre CRC\u2010MHC University Hospital of Tours  Tours France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Johann","family":"Rose","sequence":"additional","affiliation":[{"name":"Haemophilia Treatment Centre Hospital of Le Mans  Le Mans France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Philippe","family":"Beurrier","sequence":"additional","affiliation":[{"name":"Haemophilia Treatment Centre CT\u2010MHC University Hospital of Angers  Angers France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2938-8013","authenticated-orcid":false,"given":"Beno\u00eet","family":"Guillet","sequence":"additional","affiliation":[{"name":"Haemophilia Treatment Centre CRC\u2010MHC; IRSET (Research Institute for Health, Environment and Work) \u2013 UMR_S 1085, CHU Rennes Universit\u00e9 de Rennes  Rennes France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Imane","family":"Aoubiza","sequence":"additional","affiliation":[{"name":"INSERM, LTSI\u2010UM R 1099, CHU Rennes Universit\u00e9 de Rennes  Rennes France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Morgane","family":"Pierre\u2010Jean","sequence":"additional","affiliation":[{"name":"INSERM, LTSI\u2010UM R 1099, CHU Rennes Universit\u00e9 de Rennes  Rennes France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yohann","family":"Repesse","sequence":"additional","affiliation":[{"name":"Haemophilia Treatment Centre CRC\u2010MHC University Hospital of Caen Normandie  Caen France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Claire","family":"de Moreuil","sequence":"additional","affiliation":[{"name":"Internal Medicine Department University Hospital of Brest  Brest France"},{"name":"INSERM, UMR 1304\u2010GETBO Universit\u00e9 de Brest Brest France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9853-2635","authenticated-orcid":false,"given":"Brigitte","family":"Pan\u2010Petesch","sequence":"additional","affiliation":[{"name":"Haemophilia Treatment Centre CRC\u2010MHC, Haematology University Hospital of Brest  Brest France"},{"name":"INSERM, UMR 1304\u2010GETBO Universit\u00e9 de Brest Brest France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,5,31]]},"reference":[{"key":"e_1_2_13_2_1","unstructured":"Fili\u00e8re des Maladies H\u00e9morragiques Constitutionnelles (MHEMO). \u201cLa Maladie De Willebrand.\u201d Accessed May 14 2024 https:\/\/mhemo.fr\/les\u2010pathologies\/la\u2010maladie\u2010de\u2010willebrand\/."},{"key":"e_1_2_13_3_1","unstructured":"Haute Autorit\u00e9 de Sant\u00e9 (HAS). \u201cProtocole National de Diagnostic et de Soins (PNDS) Maladie de Willebrand: Argumentaire 2021.\u201d Accessed May 14 2024 https:\/\/www.has\u2010sante.fr\/upload\/docs\/application\/pdf\/2021\u201002\/maladie_de_willebrand_\u2010_argumentaire.pdf."},{"key":"e_1_2_13_4_1","doi-asserted-by":"publisher","DOI":"10.2147\/JBM.S389241"},{"key":"e_1_2_13_5_1","doi-asserted-by":"publisher","DOI":"10.1053\/j.seminhematol.2004.12.005"},{"key":"e_1_2_13_6_1","doi-asserted-by":"publisher","DOI":"10.1111\/jth.12130"},{"key":"e_1_2_13_7_1","doi-asserted-by":"publisher","DOI":"10.1111\/jth.13115"},{"key":"e_1_2_13_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.thromres.2012.06.006"},{"key":"e_1_2_13_9_1","doi-asserted-by":"publisher","DOI":"10.14503\/THIJ\u201021\u20107470"},{"key":"e_1_2_13_10_1","doi-asserted-by":"publisher","DOI":"10.1007\/s11239\u2010006\u20100002\u20109"},{"key":"e_1_2_13_11_1","doi-asserted-by":"publisher","DOI":"10.9734\/IBRR\/2014\/289"},{"key":"e_1_2_13_12_1","doi-asserted-by":"publisher","DOI":"10.1097\/MBC.0000000000000177"},{"key":"e_1_2_13_13_1","doi-asserted-by":"publisher","DOI":"10.1055\/s\u20100031\u20101317577"},{"key":"e_1_2_13_14_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12498"},{"key":"e_1_2_13_15_1","doi-asserted-by":"publisher","DOI":"10.1179\/1607845412Y.0000000059"},{"key":"e_1_2_13_16_1","doi-asserted-by":"publisher","DOI":"10.1182\/bloodadvances.2020003264"},{"key":"e_1_2_13_17_1","unstructured":"International Society on Thrombosis and Haemostasis. \u201cISTH\u2010SSC Bleeding Assessment Tool.\u201d Accessed October 15 2024 https:\/\/cdn.ymaws.com\/www.isth.org\/resource\/resmgr\/ssc\/isth\u2010ssc_bleeding_assessment.pdf."},{"key":"e_1_2_13_18_1","unstructured":"Soci\u00e9t\u00e9 fran\u00e7aise de cardiologie. \u201cFacteurs de Risque Cardiovasculaire et Pr\u00e9vention.\u201d In Coll\u00e8ge National Des Enseignants de Cardiologie 2019. Accessed May 14 2024 https:\/\/www.sfcardio.fr\/sites\/default\/files\/2019\u201011\/2015\u20102e_Ref_Cardio_ch02_facteurs_cv.pdf."},{"key":"e_1_2_13_19_1","unstructured":"Coll\u00e8ge National des Enseignants de Pneumologie. \u201cEmbolie Pulmonaire et Thrombose Veineuse Profonde: Prescription Et Surveillance Des Antithrombotiques.\u201d Accessed May 14 2024 https:\/\/cep.splf.fr\/wp\u2010content\/uploads\/2018\/09\/items_224_MTEV_2018.pdf."},{"key":"e_1_2_13_20_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1538\u20107836.2012.04769.x"},{"key":"e_1_2_13_21_1","unstructured":"World Health Organization (WHO). \u201cList of Official ICD\u201010 Updates (CIM10).\u201d Accessed October 15 2024 https:\/\/www.who.int\/standards\/classifications\/classification\u2010of\u2010diseases\/list\u2010of\u2010official\u2010icd\u201010\u2010updates."},{"key":"e_1_2_13_22_1","doi-asserted-by":"publisher","DOI":"10.2196\/50194"},{"key":"e_1_2_13_23_1","unstructured":"The R Project for Statistical Computing. \u201cR: The R Project for Statistical Computing.\u201d Accessed October 8 2024 https:\/\/www.r\u2010project.org\/."},{"key":"e_1_2_13_24_1","unstructured":"Institut de Radioprotection et de S\u00fbret\u00e9 Nucl\u00e9aire. \u201cLa Pr\u00e9vention et la Prise en Charge Des Accidents Vasculaires C\u00e9r\u00e9braux en France: Synth\u00e8se Du Rapport \u00e0 Madame la Ministre De la Sant\u00e9 et Des Sports.\u201d Accessed July 19 2024 https:\/\/sante.gouv.fr\/IMG\/pdf\/avc_\u2010_synthese_seule_rapport_final_\u2010_vf.pdf."},{"key":"e_1_2_13_25_1","unstructured":"Sant\u00e9 Publique France. \u201cInfarctus Du Myocarde.\u201d Accessed July 19 2024 https:\/\/www.santepubliquefrance.fr\/maladies\u2010et\u2010traumatismes\/maladies\u2010cardiovasculaires\u2010et\u2010accident\u2010vasculaire\u2010cerebral\/infarctus\u2010du\u2010myocarde."}],"container-title":["Haemophilia"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.70309","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/hae.70309","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.70309","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,9,19]],"date-time":"2026-09-19T09:41:35Z","timestamp":1789810895000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/hae.70309"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,5,31]]},"references-count":24,"journal-issue":{"issue":"5","published-print":{"date-parts":[[2026,9]]}},"alternative-id":["10.1111\/hae.70309"],"URL":"https:\/\/doi.org\/10.1111\/hae.70309","archive":["Portico"],"relation":{},"ISSN":["1351-8216","1365-2516"],"issn-type":[{"value":"1351-8216","type":"print"},{"value":"1365-2516","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,5,31]]},"assertion":[{"value":"2025-09-13","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-04-27","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-05-31","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}