{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,23]],"date-time":"2026-10-23T19:02:41Z","timestamp":1792782161436,"version":"4.5.0"},"reference-count":31,"publisher":"Wiley","issue":"12","license":[{"start":{"date-parts":[[2015,11,17]],"date-time":"2015-11-17T00:00:00Z","timestamp":1447718400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"name":"Dutch Hemophilia Foundation"},{"name":"CSL Behring"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["American J Hematol"],"published-print":{"date-parts":[[2015,12]]},"abstract":"<jats:p>\n                    The bleeding phenotype of children with von Willebrand disease (VWD) needs to be characterized in detail to facilitate diagnosis during childhood and aid in the planning and assessment of treatment strategies. The objective was to evaluate the occurrence, type, and severity of bleeding in a large cohort of children with moderate and severe VWD. We included 113 children (aged 0\u201316 years) with Type 1 (\n                    <jats:italic>n<\/jats:italic>\n                    \u2009=\u200960), 2 (\n                    <jats:italic>n<\/jats:italic>\n                    \u2009=\u200944), and 3 (\n                    <jats:italic>n<\/jats:italic>\n                    \u2009=\u20099) VWD with von Willebrand factor (VWF) antigen and\/or VWF ristocetin cofactor levels\u2009\u2264\u200930 U\/dL from a nation\u2010wide cross\u2010sectional study (\u201cWillebrand in the Netherlands\u201d study). Bleeding severity and frequency were determined using the International Society on Thrombosis and Hemostasis\u2010Bleeding Assessment Tool (ISTH\u2010BAT) with supplementary pediatric\u2010specific bleeding symptoms (umbilical stump bleeding, cephalohematoma, cheek hematoma, conjunctival bleeding, postcircumcision and postvenipuncture bleeding). We found that all 26 postmenarche girls experienced menorrhagia. Other common bleedings were cutaneous (81%), oropharyngeal (64%), prolonged bleeding from minor wounds (58%), and epistaxis (56%). Pediatric\u2010specific bleeding symptoms were present in 44% of patients. ISTH\u2010BAT bleeding score was higher in index cases than in affected family members (median, 12.0 vs. 6.5,\n                    <jats:italic>P<\/jats:italic>\n                    \u2009&lt;\u20090.001), higher in Type 3 VWD than in Type 2 or 1 (17.0 vs. 10.5 or 6.5,\n                    <jats:italic>P<\/jats:italic>\n                    \u2009&lt;\u20090.001) and higher in children with severe (&lt;10 U\/dL) than moderate VWD (10\u201330 U\/dL) (11.0 vs. 7.0,\n                    <jats:italic>P<\/jats:italic>\n                    \u2009&lt;\u20090.001). Frequency of any bleeding, epistaxis, and oral cavity was higher in types 2 and 3 than in Type 1 VWD and was associated with VWF levels. We conclude that pediatric\u2010specific bleeding symptoms occurred in a large proportion of children with moderate or severe VWD and should be included when evaluating children for VWD. Am. J. Hematol. 90:1142\u20131148, 2015. \u00a9 2015 Wiley Periodicals, Inc.\n                  <\/jats:p>","DOI":"10.1002\/ajh.24195","type":"journal-article","created":{"date-parts":[[2015,9,16]],"date-time":"2015-09-16T19:44:45Z","timestamp":1442432685000},"page":"1142-1148","source":"Crossref","is-referenced-by-count":61,"title":["Bleeding spectrum in children with moderate or severe von\n                    <scp>W<\/scp>\n                    illebrand disease:\n                    <scp>R<\/scp>\n                    elevance of pediatric\u2010specific bleeding"],"prefix":"10.1002","volume":"90","author":[{"given":"Yvonne V.","family":"Sanders","sequence":"first","affiliation":[{"name":"Department of Hematology Erasmus University Medical Center Rotterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Karin","family":"Fijnvandraat","sequence":"additional","affiliation":[{"name":"Department of Pediatric Hematology Emma Children's Hospital, Academic Medical Center Amsterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Johan","family":"Boender","sequence":"additional","affiliation":[{"name":"Department of Hematology Erasmus University Medical Center Rotterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Evelien P.","family":"Mauser\u2010Bunschoten","sequence":"additional","affiliation":[{"name":"Van Creveldkliniek\/Department of Benign Hematology University Medical Center Utrecht Utrecht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Johanna G.","family":"van der Bom","sequence":"additional","affiliation":[{"name":"Department of Clinical Epidemiology Leiden University Medical Center Leiden The Netherlands"},{"name":"Jon J Van Rood Center for Clinical Transfusion Medicine, Sanquin Research Leiden The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joke","family":"de Meris","sequence":"additional","affiliation":[{"name":"Netherlands Hemophilia Society Leiden The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frans J.","family":"Smiers","sequence":"additional","affiliation":[{"name":"Department of Pediatric Hematology Willem\u2010Alexander Children's Hospital, Leiden University Medical Center Leiden The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Bernd","family":"Granzen","sequence":"additional","affiliation":[{"name":"Department of Pediatric Hematology Maastricht University Medical Center Maastricht The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Paul","family":"Brons","sequence":"additional","affiliation":[{"name":"Department of Pediatric Hematology Radboud University Medical Center Nijmegen The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rienk Y.J.","family":"Tamminga","sequence":"additional","affiliation":[{"name":"Department of Pediatric Hematology University Medical Center Groningen Groningen The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Marjon H.","family":"Cnossen","sequence":"additional","affiliation":[{"name":"Department of Pediatric Hematology Erasmus University Medical Center\u2010Sophia Children's Hospital Rotterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Frank W.G.","family":"Leebeek","sequence":"additional","affiliation":[{"name":"Department of Hematology Erasmus University Medical Center Rotterdam The Netherlands"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"name":"the WiN Study 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