{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,19]],"date-time":"2026-09-19T12:14:53Z","timestamp":1789820093463,"version":"4.0.1"},"reference-count":19,"publisher":"Wiley","issue":"4","license":[{"start":{"date-parts":[[2025,5,27]],"date-time":"2025-05-27T00:00:00Z","timestamp":1748304000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"},{"start":{"date-parts":[[2025,5,27]],"date-time":"2025-05-27T00:00:00Z","timestamp":1748304000000},"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":[[2025,7]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Introduction<\/jats:title>\n                    <jats:p>About 40%\u201370% of persons with a clinically relevant bleeding tendency who are referred to haemostasis experts are classified as having a \u2018bleeding disorder of unknown cause\u2019 (BDUC) as no biological entity can be found after extensive laboratory testing. Currently, guidelines are under development regarding diagnostic assessment and management to minimize variation in clinical practice.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Aim<\/jats:title>\n                    <jats:p>Investigate current practices regarding BDUC in the Netherlands.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>An online survey on the best BDUC definition, associated bleeding phenotype, clinical and diagnostic approaches, treatment, registration, and follow\u2010up was distributed amongst healthcare providers working in Dutch haemophilia treatment centres (HTCs).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>The survey was completed by 39\/54 (72%) respondents. Twenty percent did not register BDUC patients in their HTC. Healthcare professionals indicated that follow\u2010up should depend on bleeding phenotype severity and bleeding history, and other potential causes for an increased bleeding tendency should be excluded. Moreover, the use of laboratory tests within the routine diagnostic pathway was demonstrated to be heterogeneous. Regarding treatment, tranexamic acid was most frequently prescribed for minor and major surgical interventions (79% and 86%), dental extractions (93%) and childbirth (93%). Desmopressin was prescribed for major surgical procedures by 79%.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>Our survey shows that Dutch current practice varies but is generally in line with recent ISTH SSC recommendations. Additionally, it describes other clinically relevant topics not included in the international survey, such as follow\u2010up and exclusion of other causes for bleeding. This survey therefore adds to international efforts to unify BDUC definition, diagnostic approach, treatment and follow\u2010up, and to attain broadly supported guidelines.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1111\/hae.70065","type":"journal-article","created":{"date-parts":[[2025,5,27]],"date-time":"2025-05-27T08:26:29Z","timestamp":1748334389000},"page":"752-760","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":5,"title":["Current Practice Regarding Bleeding Disorders of Unknown Cause in the Netherlands: A National Survey"],"prefix":"10.1111","volume":"31","author":[{"ORCID":"https:\/\/orcid.org\/0009-0009-8970-9473","authenticated-orcid":false,"given":"Caroline M. A.","family":"Mussert","sequence":"first","affiliation":[{"name":"Department of Pediatric Hematology and Oncology Erasmus MC Sophia Children's Hospital University Medical Center Rotterdam  Rotterdam the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0007-0208-5764","authenticated-orcid":false,"given":"Amaury L. L.","family":"Monard","sequence":"additional","affiliation":[{"name":"Department of Hematology Maastricht University Medical Center  Maastricht the Netherlands"},{"name":"CARIM \u2013 School for Cardiovascular Disease Maastricht University  Maastricht the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Tirsa T.","family":"van Duijl","sequence":"additional","affiliation":[{"name":"Department of Molecular Hematology Sanquin Research  Amsterdam the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yvonne M. C.","family":"Henskens","sequence":"additional","affiliation":[{"name":"CARIM \u2013 School for Cardiovascular Disease Maastricht University  Maastricht the Netherlands"},{"name":"Department of Central Diagnostic Laboratory Maastricht University Medical Center  Maastricht the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maartje","family":"van den Biggelaar","sequence":"additional","affiliation":[{"name":"Department of Molecular Hematology Sanquin Research  Amsterdam the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2762-6033","authenticated-orcid":false,"given":"Roger E. G.","family":"Schutgens","sequence":"additional","affiliation":[{"name":"Center for Benign Haematology Thrombosis and Haemostasis Van Creveldkliniek University Medical Center Utrecht  Utrecht the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Saskia E. M.","family":"Schols","sequence":"additional","affiliation":[{"name":"Department Hematology Radboud University Medical Center the Netherlands\u00a0and Hemophilia Treatment Centre Nijmegen\u2010Eindhoven\u2010Maastricht  Nijmegen the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karin J.","family":"Fijnvandraat","sequence":"additional","affiliation":[{"name":"Department Pediatric Hematology Amsterdam UMC Emma Children's Hospital University of Amsterdam  Amsterdam the Netherlands"},{"name":"Department of Molecular Cellular Hemostasis Sanquin Research  Amsterdam the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karina","family":"Meijer","sequence":"additional","affiliation":[{"name":"Department Hematology University Medical Center Groningen  Groningen the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paul L.","family":"den Exter","sequence":"additional","affiliation":[{"name":"Department Vascular Medicine Leiden University Medical Center  Leiden the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Laurens","family":"Nieuwenhuizen","sequence":"additional","affiliation":[{"name":"Department Internal Medicine Maxima Medical Center  Veldhoven the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0248-4710","authenticated-orcid":false,"given":"Iris","family":"van Moort","sequence":"additional","affiliation":[{"name":"Department Hematology Erasmus MC University Medical Center Rotterdam  Rotterdam the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0265-4871","authenticated-orcid":false,"given":"Marieke J. H. A.","family":"Kruip","sequence":"additional","affiliation":[{"name":"Department Hematology Erasmus MC University Medical Center Rotterdam  Rotterdam the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marjon H.","family":"Cnossen","sequence":"additional","affiliation":[{"name":"Department of Pediatric Hematology and Oncology Erasmus MC Sophia Children's Hospital University Medical Center Rotterdam  Rotterdam the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3281-926X","authenticated-orcid":false,"given":"Floor C. J. I.","family":"Heubel\u2010Moenen","sequence":"additional","affiliation":[{"name":"Department of Hematology Maastricht University Medical Center  Maastricht the Netherlands"},{"name":"CARIM \u2013 School for Cardiovascular Disease Maastricht University  Maastricht the Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"for the BDUC\u2010iN study group","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2025,5,27]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.14174"},{"key":"e_1_2_10_3_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jtha.2024.03.005"},{"key":"e_1_2_10_4_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13422"},{"key":"e_1_2_10_5_1","doi-asserted-by":"publisher","DOI":"10.1111\/ijlh.13124"},{"key":"e_1_2_10_6_1","doi-asserted-by":"publisher","DOI":"10.3324\/haematol.10816"},{"key":"e_1_2_10_7_1","unstructured":"SurveyMonkey. \u201cThe World's Most Popular Survey Platform\u201d1999 https:\/\/www.surveymonkey.com\/."},{"key":"e_1_2_10_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jtha.2023.03.006"},{"key":"e_1_2_10_9_1","doi-asserted-by":"publisher","DOI":"10.1097\/HS9.0000000000000286"},{"key":"e_1_2_10_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/bjh.18003"},{"key":"e_1_2_10_11_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jtha.2025.01.011"},{"key":"e_1_2_10_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jtha.2024.05.027"},{"key":"e_1_2_10_13_1","doi-asserted-by":"publisher","DOI":"10.1097\/MBC.0000000000000444"},{"key":"e_1_2_10_14_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.15059"},{"key":"e_1_2_10_15_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00277-011-1201-8"},{"key":"e_1_2_10_16_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00277-016-2893-6"},{"key":"e_1_2_10_17_1","doi-asserted-by":"publisher","DOI":"10.1111\/jth.14529"},{"key":"e_1_2_10_18_1","doi-asserted-by":"publisher","DOI":"10.1002\/rth2.12681"},{"key":"e_1_2_10_19_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.13991"},{"key":"e_1_2_10_20_1","doi-asserted-by":"publisher","DOI":"10.1111\/hae.12811"}],"container-title":["Haemophilia"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.70065","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/hae.70065","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/hae.70065","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,3,8]],"date-time":"2026-03-08T20:34:50Z","timestamp":1773002090000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/hae.70065"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,5,27]]},"references-count":19,"journal-issue":{"issue":"4","published-print":{"date-parts":[[2025,7]]}},"alternative-id":["10.1111\/hae.70065"],"URL":"https:\/\/doi.org\/10.1111\/hae.70065","archive":["Portico"],"relation":{},"ISSN":["1351-8216","1365-2516"],"issn-type":[{"value":"1351-8216","type":"print"},{"value":"1365-2516","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,5,27]]},"assertion":[{"value":"2025-02-14","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-05-13","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2025-05-27","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}