Show simple item record

dc.contributor.authorO’Hara, Jamie*
dc.contributor.authorWalsh, Shaun*
dc.contributor.authorCamp, Charlotte*
dc.contributor.authorMazza, Giuseppe*
dc.contributor.authorCarroll, Liz*
dc.contributor.authorHoxer, Christina*
dc.contributor.authorWilkinson, Lars*
dc.date.accessioned2018-06-07T12:05:18Z
dc.date.available2018-06-07T12:05:18Z
dc.date.issued2018-05-02
dc.identifier.citationO’Hara, J., et el. (2018). The impact of severe haemophilia and the presence of target joints on health-related quality-of-life. Health and Quality of Life Outcomes, 16(1), 84. http://doi.org/10.1186/s12955-018-0908-9
dc.identifier.doi10.1186/s12955-018-0908-9
dc.identifier.urihttp://hdl.handle.net/10034/621173
dc.description.abstractBackground: Joint damage remains a major complication associated with haemophilia and is widely accepted as one of the most debilitating symptoms for persons with severe haemophilia. The aim of this study is to describe how complications of haemophilia such as target joints influence health-related quality of life (HRQOL). Methods: Data on hemophilia patients without inhibitors were drawn from the ‘Cost of Haemophilia across Europe – a Socioeconomic Survey’ (CHESS) study, a cost-of-illness assessment in severe haemophilia A and B across five European countries (France, Germany, Italy, Spain, and the UK). Physicians provided clinical and sociodemographic information for 1285 adult patients, 551 of whom completed corresponding questionnaires, including EQ-5D. A generalised linear model was developed to investigate the relationship between EQ-5D index score and target joint status (defined in the CHESS study as areas of chronic synovitis), adjusted for patient covariates including socio-demographic characteristics and comorbidities. Results: Five hundred and fifteen patients (42% of the sample) provided an EQ-5D response; a total of 692 target joints were recorded across the sample. Mean EQ-5D index score for patients with no target joints was 0.875 (standard deviation [SD] 0.179); for patients with one or more target joints, mean index score was 0.731 (SD 0.285). Compared to having no target joints, having one or more target joints was associated with lower index scores (average marginal effect (AME) -0.120; SD 0.0262; p < 0.000). Conclusions: This study found that the presence of chronic synovitis has a significant negative impact on HRQOL for adults with severe haemophilia. Prevention, early diagnosis and treatment of target joints should be an important consideration for clinicians and patients when managing haemophilia.
dc.language.isoenen
dc.publisherBioMed Centralen
dc.relation.urlhttps://hqlo.biomedcentral.com/articles/10.1186/s12955-018-0908-9en
dc.subjectHaemophiliaen
dc.subjectDisease burdenen
dc.subjectJoint diseaseen
dc.subjectHealth-related quality of lifeen
dc.subjectPatient-reported outcome measuresen
dc.titleThe impact of severe haemophilia and the presence of target joints on health-related quality-of-lifeen
dc.typeArticleen
dc.identifier.eissn1477-7525
dc.contributor.departmentUniversity of Chester; HCD Economics; University College London; The Haemophilia Society; Novo Nordisk
dc.identifier.journalHealth and Quality of Life Outcomes
dc.language.rfc3066en
dc.rights.holderThe Author(s).
dc.date.updated2018-06-01T11:40:45Z
dc.date.accepted2018-04-20
or.grant.openaccessYesen
rioxxterms.funderNovo Nordisken
rioxxterms.identifier.projectNovo Nordisken
rioxxterms.versionVoRen
rioxxterms.licenseref.startdate2018-05-02
refterms.dateFCD2019-07-16T13:37:00Z
refterms.versionFCDVoR
refterms.dateFOA2018-08-13T14:18:08Z
html.description.abstractBackground: Joint damage remains a major complication associated with haemophilia and is widely accepted as one of the most debilitating symptoms for persons with severe haemophilia. The aim of this study is to describe how complications of haemophilia such as target joints influence health-related quality of life (HRQOL). Methods: Data on hemophilia patients without inhibitors were drawn from the ‘Cost of Haemophilia across Europe – a Socioeconomic Survey’ (CHESS) study, a cost-of-illness assessment in severe haemophilia A and B across five European countries (France, Germany, Italy, Spain, and the UK). Physicians provided clinical and sociodemographic information for 1285 adult patients, 551 of whom completed corresponding questionnaires, including EQ-5D. A generalised linear model was developed to investigate the relationship between EQ-5D index score and target joint status (defined in the CHESS study as areas of chronic synovitis), adjusted for patient covariates including socio-demographic characteristics and comorbidities. Results: Five hundred and fifteen patients (42% of the sample) provided an EQ-5D response; a total of 692 target joints were recorded across the sample. Mean EQ-5D index score for patients with no target joints was 0.875 (standard deviation [SD] 0.179); for patients with one or more target joints, mean index score was 0.731 (SD 0.285). Compared to having no target joints, having one or more target joints was associated with lower index scores (average marginal effect (AME) -0.120; SD 0.0262; p < 0.000). Conclusions: This study found that the presence of chronic synovitis has a significant negative impact on HRQOL for adults with severe haemophilia. Prevention, early diagnosis and treatment of target joints should be an important consideration for clinicians and patients when managing haemophilia.


Files in this item

Thumbnail
Name:
12955_2018_Article_908.pdf
Size:
789.2Kb
Format:
PDF

This item appears in the following Collection(s)

Show simple item record