ArticleThe European journal of health economics : HEPAC : health economics in prevention and care2016
Social/economic costs and quality of life in patients with haemophilia in Europe.
Article in The European journal of health economics : HEPAC : health economics in prevention and care, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 35 papers, 2 of them syntheses that pooled it.
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The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
35 citing papers in PubMed, 2 syntheses or guidelines pooled it, 79 citations in OpenAlex.
- Cost-of-illness studies in nine Central and Eastern European countries.The European journal of health economics : HEPAC : health economics in prevention and care · 2019Pooled it
- Spillover Effects on Caregivers' and Family Members' Utility: A Systematic Review of the Literature.PharmacoEconomics · 2019Pooled it
- Effect of emicizumab prophylaxis on bone and joint health markers in people with haemophilia A without factor VIII inhibitors in the HAVEN 3 study.Haemophilia : the official journal of the World Federation of Hemophilia · 2022Trial
- Effectiveness of hypnosis for pain management and promotion of health-related quality-of-life among people with haemophilia: a randomised controlled pilot trial.Scientific reports · 2019Trial
- Health-related quality of life in paediatric haemophilia B patients treated with rIX-FP.Haemophilia : the official journal of the World Federation of Hemophilia · 2019Trial
- Psychosocial Outcomes and Quality of Life in Patients with Hemophilia a Without Inhibitors: The HemoLIFE Study.Journal of clinical medicine · 2026Article
- Structural Enablers of Rare Disease Treatment Coverage in Latin America and the Caribbean: Lessons from Emicizumab.Journal of market access & health policy · 2026Article
- Clinical, humanistic, and economic burden of haemophilia A in China: findings from a real-world survey.Orphanet journal of rare diseases · 2025Article
- Economic Burden of Haemophilia from a Societal Perspective: A Scoping Review.PharmacoEconomics - open · 2025Review
- Distribution and predictors of haemophilia-related costs in the United Kingdom: analysis of the CHESS I and CHESS II burden of illness studies.BMC health services research · 2024Article
- On spillovers in economic evaluations: definition, mapping review and research agenda.The European journal of health economics : HEPAC : health economics in prevention and care · 2024Review
- Shortage of plasma-derived medicinal products: what is next? narrative literature review on its causes and counteracting policies in Italy.Frontiers in pharmacology · 2024Review
- Health-related quality of life in patients with diverse rare diseases: An online survey.Genetics in medicine open · 2024Article
- Methods used to account for caregivers' sex and gender within studies examining the financial burden of caregivers of children and adolescents : Results from a scoping review.ClinicoEconomics and outcomes research : CEOR · 2024Article
- Are We Capturing the Socioeconomic Burden of Rare Genetic Disease? A Scoping Review of Economic Evaluations and Cost-of-Illness Studies.PharmacoEconomics · 2023Article
- Cost-effectiveness of recombinant factor VIII Fc versus emicizumab for prophylaxis in adults and adolescents with haemophilia A without inhibitors in the UK.European journal of haematology · 2023Article
- Area under the curve: Comparing the value of factor VIII replacement therapies in haemophilia A.Haemophilia : the official journal of the World Federation of Hemophilia · 2023Article
- Rare disease curative care expenditure-financing scheme-health provider-beneficiary group analysis: an empirical study in Sichuan Province, China.Orphanet journal of rare diseases · 2022Article
- Health-related quality of life, direct medical and societal costs among children with moderate or severe haemophilia in Europe: multivariable models of the CHESS-PAEDs study.Orphanet journal of rare diseases · 2022Article
- Differential humanistic and economic burden of mild, moderate and severe haemophilia in european adults: a regression analysis of the CHESS II study.Orphanet journal of rare diseases · 2022Article
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
19 authors at 12 institutions in 8 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
objectiveThe aim of this study was to determine the economic burden from a societal perspective and the health-related quality of life (HRQOL) of patients with haemophilia in Europe.
methodsWe conducted a cross-sectional study of patients with haemophilia from Bulgaria, France, Germany, Hungary, Italy, Spain Sweden and the UK. Data on demographic characteristics, health resource utilisation, informal care, loss of labour productivity and HRQOL were collected from the questionnaires completed by patients or their caregivers. HRQOL was measured with the EuroQol 5-domain (EQ-5D) questionnaire. The costs have been estimated from a societal perspective adopting a bottom-up approach.
resultsA total of 401 questionnaires were included in the study, of which 339 were collected from patients with haemophilia and 62 from caregivers. The lowest average annual cost per person was reported in Bulgaria (€6,660) and the highest in Germany (€194,490). Our results demonstrate both a large difference from country to country in the average annual cost per patient in 2012 and the driving role of drugs in costs. Drugs represent nearly 90 % of direct healthcare costs in a majority of the countries analysed (Hungary, Italy, Spain and Germany). In Bulgaria, France and Sweden, however, healthcare services (visits, tests and hospitalisations) prevail. Costs are also shown to differ between children and adults. The mean EQ-5D index score for adult patients was 0.69 and mean EQ-5D VAS was 66.6. The mean EQ-5D index score for carers was 0.87 and mean EQ-5D VAS was 75.5. In the disability score, 60 % showed no disability and measuring caregiver burden with the Zarit Index produced an overall mean score of 25.3.
conclusionWe have shown that haemophilia is associated with a substantial economic burden and impaired HRQOL. Studies on cost of illness and HRQOL are important for haemophilia as the future of this disease is likely to change with the development of new innovative treatments. The introduction of these treatments will most likely impact future costs related to haemophilia.
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