{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,1,10]],"date-time":"2026-01-10T09:28:08Z","timestamp":1768037288410,"version":"3.49.0"},"reference-count":60,"publisher":"Wiley","issue":"2","license":[{"start":{"date-parts":[[2022,1,18]],"date-time":"2022-01-18T00:00:00Z","timestamp":1642464000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0\/"}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Haemophilia"],"published-print":{"date-parts":[[2022,3]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Aim<\/jats:title><jats:p>Currently, it is unknown which patient\u2010reported outcomes are important for patients with autosomal inherited bleeding disorders. Therefore, the purpose of this study is to systematically review the available literature assessing patient\u2010reported outcomes and their measurement methods in autosomal inherited bleeding disorders.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>The Embase, Medline ALL, Web of Science Core Collection, Cochrane Central Register of Controlled Trails and Google Scholar databases were searched from inception until 14 August 2020. Studies on patient\u2010reported outcomes in patients with von Willebrand disease, inherited platelet function disorders and coagulation factor deficiencies were included.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Twenty\u2010one articles met the inclusion criteria. Three studies were assessed as having poor quality, and therefore a high risk of bias. Nineteen studies had fair quality rating. Different measurements methods were used, ranging from predefined to self\u2010developed questionnaires. The majority of included studies focused on von Willebrand disease. Patients with von Willebrand disease reported lower health\u2010related quality of life compared to the general population. Overall, this trend was especially visible in the following domains: vitality, physical and social functioning and pain. Women with inherited bleeding disorders scored lower on health\u2010related quality of life compared to men, especially women with heavy menstrual bleeding. Patients with joint bleeds or heavy menstrual bleeding reported an increased level of pain.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>Patients with autosomal inherited bleeding disorders report lower health related quality of life, especially those with joint bleeds or heavy menstrual bleeding. Numerous measurement methods are used in patients with autosomal inherited bleeding disorders, highlighting the need for studies using established, standardized measurement methods.<\/jats:p><\/jats:sec>","DOI":"10.1111\/hae.14492","type":"journal-article","created":{"date-parts":[[2022,1,18]],"date-time":"2022-01-18T04:41:47Z","timestamp":1642480907000},"page":"197-214","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":11,"title":["Patient\u2010reported outcomes in autosomal inherited bleeding disorders: A systematic literature review"],"prefix":"10.1111","volume":"28","author":[{"given":"Evelien S.","family":"van Hoorn","sequence":"first","affiliation":[{"name":"Erasmus MC University Medical Centre Rotterdam, Department of Public Health  Rotterdam The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Maite E.","family":"Houwing","sequence":"additional","affiliation":[{"name":"Erasmus MC \u2013 Sophia Children's Hospital University Medical Centre Rotterdam, Department of Paediatric Haematology  Rotterdam The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Wala","family":"Al Arashi","sequence":"additional","affiliation":[{"name":"Erasmus MC \u2013 Sophia Children's Hospital University Medical Centre Rotterdam, Department of Paediatric Haematology  Rotterdam The Netherlands"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Frank W. 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