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dc.contributor.authorO’Hara, Jamieen
dc.contributor.authorWalsh, Shaunen
dc.contributor.authorCamp, Charlotteen
dc.contributor.authorMazza, Giuseppeen
dc.contributor.authorCarroll, Lizen
dc.contributor.authorHoxer, Christinaen
dc.contributor.authorWilkinson, Larsen
dc.date.accessioned2018-02-02T12:01:32Z
dc.date.available2018-02-02T12:01:32Z
dc.date.issued2018-01-16
dc.identifier.citationO’Hara, J., et al. (2018). The relationship between target joints and direct resource use in severe haemophilia. Health Economics Review, 8(1). https://doi.org/10.1186/s13561-018-0185-7en
dc.identifier.doi10.1186/s13561-018-0185-7
dc.identifier.urihttp://hdl.handle.net/10034/620848
dc.description.abstractObjectives Target joints are a common complication of severe haemophilia. While factor replacement therapy constitutes the majority of costs in haemophilia, the relationship between target joints and non drug-related direct costs (NDDCs) has not been studied. Methods Data on haemophilia patients without inhibitors was drawn from the ‘Cost of Haemophilia across Europe – a Socioeconomic Survey’ (CHESS) study, a cost assessment in severe haemophilia A and B across five European countries (France, Germany, Italy, Spain, and the United Kingdom) in which 139 haemophilia specialists provided demographic and clinical information for 1285 adult patients. NDDCs were calculated using publicly available cost data, including 12-month ambulatory and secondary care activity: haematologist and other specialist consultant consultations, medical tests and examinations, bleed-related hospital admissions, and payments to professional care providers. A generalized linear model was developed to investigate the relationship between NDDCs and target joints (areas of chronic synovitis), adjusted for patient covariates. Results Five hundred and thirteen patients (42% of the sample) had no diagnosed target joints; a total of 1376 target joints (range 1–10) were recorded in the remaining 714 patients. Mean adjusted NDDCs for persons with no target joints were EUR 3134 (standard error (SE) EUR 158); for persons with one or more target joints, mean adjusted NDDCs were EUR 3913 (SE EUR 157; average mean effect EUR 779; p < 0.001). Conclusions Our analysis suggests that the presence of one or more target joints has a significant impact on NDDCs for patients with severe haemophilia, ceteris paribus. Prevention and management of target joints should be an important consideration of managing haemophilia patients.
dc.language.isoenen
dc.publisherSpringerOpenen
dc.rights.urihttp://creativecommons.org/licenses/by/4.0/en
dc.subjectHaemophiliaen
dc.subjectCost of illnessen
dc.subjectTarget jointsen
dc.subjectBurden of diseaseen
dc.subjectArthorapthyen
dc.subjectSynovitisen
dc.titleThe relationship between target joints and direct resource use in severe haemophiliaen
dc.typeArticleen
dc.identifier.eissn2191-1991
dc.contributor.departmentUniversity of Chester; HCD Economics, The Innovation Centre, Daresbury; University College London; The Haemophilia Society; Novo Nordisk A/Sen
dc.identifier.journalHealth Economics Review
dc.language.rfc3066en
dc.rights.holderThe Author(s).
dc.date.updated2018-02-01T04:39:07Z
dc.date.accepted2018-01-03
or.grant.openaccessYesen
rioxxterms.funderNovo Nordisken
rioxxterms.identifier.projectNovo Nordisken
rioxxterms.versionVoRen
rioxxterms.licenseref.startdate2018-01-16
refterms.dateFOA2018-08-13T21:23:28Z
html.description.abstractObjectives Target joints are a common complication of severe haemophilia. While factor replacement therapy constitutes the majority of costs in haemophilia, the relationship between target joints and non drug-related direct costs (NDDCs) has not been studied. Methods Data on haemophilia patients without inhibitors was drawn from the ‘Cost of Haemophilia across Europe – a Socioeconomic Survey’ (CHESS) study, a cost assessment in severe haemophilia A and B across five European countries (France, Germany, Italy, Spain, and the United Kingdom) in which 139 haemophilia specialists provided demographic and clinical information for 1285 adult patients. NDDCs were calculated using publicly available cost data, including 12-month ambulatory and secondary care activity: haematologist and other specialist consultant consultations, medical tests and examinations, bleed-related hospital admissions, and payments to professional care providers. A generalized linear model was developed to investigate the relationship between NDDCs and target joints (areas of chronic synovitis), adjusted for patient covariates. Results Five hundred and thirteen patients (42% of the sample) had no diagnosed target joints; a total of 1376 target joints (range 1–10) were recorded in the remaining 714 patients. Mean adjusted NDDCs for persons with no target joints were EUR 3134 (standard error (SE) EUR 158); for persons with one or more target joints, mean adjusted NDDCs were EUR 3913 (SE EUR 157; average mean effect EUR 779; p < 0.001). Conclusions Our analysis suggests that the presence of one or more target joints has a significant impact on NDDCs for patients with severe haemophilia, ceteris paribus. Prevention and management of target joints should be an important consideration of managing haemophilia patients.


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