{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,10]],"date-time":"2026-10-10T18:37:42Z","timestamp":1791657462220,"version":"4.3.4"},"reference-count":32,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2024,11,27]],"date-time":"2024-11-27T00:00:00Z","timestamp":1732665600000},"content-version":"vor","delay-in-days":26,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Haemophilia"],"published-print":{"date-parts":[[2024,11]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Introduction<\/jats:title><jats:p>The needs of haemophilia carriers (HC) have been historically overlooked. It is now recognised that HC manifests bleeding symptoms, including haemarthrosis. The natural history of joint health in HC is not yet defined.<\/jats:p><\/jats:sec><jats:sec><jats:title>Aim<\/jats:title><jats:p>A multi\u2010institutional cross\u2010sectional study aimed to evaluate the characteristics of joint disease in HC, aged 18\u201340 years, compared to age\u2010matched controls.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>The carrier cohort included females with confirmed HC status. Controls had no personal or family history of bleeding disorders. All females with a history of joint trauma or surgery within 12 months or any history of joint replacement were excluded. Joint health was assessed by clinical history, Haemophilia Joint Health Score (HJHS) and point\u2010of\u2010care musculoskeletal ultrasonography (POC\u2010MSKUS).<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Thirty HC and 30 controls were enrolled. For HC, the median factor activity level was 52% (range 17%\u2013100%). Carriers, regardless of baseline factor activity levels, reported higher prevalence of chronic joint pain (<jats:italic>p<\/jats:italic>\u00a0&lt;\u00a0.001) and swelling (<jats:italic>p<\/jats:italic>\u00a0=\u00a0.002) than controls. Heavy menstrual bleeding, epistaxis, gingival bleeding and easy bruising were also more prevalent in HC (<jats:italic>p<\/jats:italic>\u00a0&lt;\u00a0.001). Despite HC having a higher median HJHS score (5\u00a0vs. 0, <jats:italic>p<\/jats:italic>\u00a0&lt;\u00a0.001), no differences were observed when using POC\u2010MSKUS. HC with a body mass index \u226525\u00a0mg\/m<jats:sup>2<\/jats:sup> reported more haemarthrosis (<jats:italic>p<\/jats:italic>\u00a0=\u00a0.037).<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusions<\/jats:title><jats:p>HC are at increased risk of joint\u2010related symptoms and poorer joint health than age\u2010matched controls. Dedicated follow\u2010up to prevent and treat joint disease in HC is imperative. This study is also a call for additional investigation to clarify the association, or lack thereof, between factor activity and joint disease.<\/jats:p><\/jats:sec>","DOI":"10.1111\/hae.15128","type":"journal-article","created":{"date-parts":[[2024,11,28]],"date-time":"2024-11-28T05:34:27Z","timestamp":1732772067000},"page":"1422-1428","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":8,"title":["Assessment of joint health in females with haemophilia: The carriers ultrasound project (CUP) study"],"prefix":"10.1111","volume":"30","author":[{"given":"Fernando F.","family":"Corrales\u2010Medina","sequence":"first","affiliation":[{"name":"Division of Pediatric Hematology\/Oncology, Department of Pediatrics University of Miami\u2010Miller School of Medicine Miami Florida USA"},{"name":"University of Miami\u2010Hemophilia Treatment Center Miami Florida USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kelli","family":"Fraga","sequence":"additional","affiliation":[{"name":"University of Miami\u2010Hemophilia Treatment Center Miami Florida USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maria","family":"D'Almeida Bastos","sequence":"additional","affiliation":[{"name":"Department of Radiology University of Miami\u2010Miller School of Medicine Miami Florida USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Amina","family":"Rafique","sequence":"additional","affiliation":[{"name":"Department of Pediatrics, Division of Pediatric Hematology\/Oncology, Levine Cancer Institute\/Atrium Health Wake Forest University School of Medicine New Orleans Louisiana USA"},{"name":"Division of Pediatric Hematology\/Oncology Tulane University School of Medicine New Orleans Louisiana USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2391-2016","authenticated-orcid":false,"given":"Christine L.","family":"Kempton","sequence":"additional","affiliation":[{"name":"Hemophilia of Georgia Center of Bleeding and Clotting Disorders Emory University Atlanta Georgia USA"},{"name":"Department of Hematology and Medical Oncology Emory University School of Medicine Atlanta Georgia USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pooja","family":"Vijayvargia","sequence":"additional","affiliation":[{"name":"Hemophilia of Georgia Center of Bleeding and Clotting Disorders Emory University Atlanta Georgia USA"},{"name":"Department of Hematology and Medical Oncology Emory University School of Medicine Atlanta Georgia USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Joanna A.","family":"Davis","sequence":"additional","affiliation":[{"name":"Division of Pediatric Hematology\/Oncology, Department of Pediatrics University of Miami\u2010Miller School of Medicine Miami Florida USA"},{"name":"University of Miami\u2010Hemophilia Treatment Center Miami Florida USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7537-0872","authenticated-orcid":false,"given":"Rachel S.","family":"Kronenfeld","sequence":"additional","affiliation":[{"name":"Division of Pediatric Hematology\/Oncology, Department of Pediatrics University of Miami\u2010Miller School of Medicine Miami Florida USA"},{"name":"University of Miami\u2010Hemophilia Treatment Center Miami Florida 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