Abstract
ObjectivesTarget 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.MethodsData 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.ResultsFive 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).ConclusionsOur 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.
Highlights
Haemophilia is an inherited, lifelong bleeding disorder characterised by prolonged traumatic or spontaneous bleeding due to a lack of clotting factor in the body
The objective of this paper is to explore the relationship between target joints and direct medical costs for persons with severe haemophilia, and the extent to which health resource utilisation and direct medical costs in severe haemophilia are driven by long-term clinical complications of the disease
Non drug-related direct costs (NDDCs) were an amalgam of 12-month ambulatory and secondary care costs gathered within the CHESS study, incorporating: haematologist and other specialist consultant consultations, medical tests and examinations, surgeries relating to joint damage, bleed-related hospital admissions, and payments to professional care providers [15]
Summary
Haemophilia is an inherited, lifelong bleeding disorder characterised by prolonged traumatic or spontaneous bleeding due to a lack of clotting factor in the body. Haemophilia is a recessive X-linked disorder and primarily affects males; symptoms are present from infancy [1]. Bleed events may be musculoskeletal or mucosal in nature but are most commonly observed in the joints of the body. In the absence of preventative ‘prophylaxis’ factor replacement therapy, most persons with severe haemophilia (
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