Observational studyBMC health services research2015
Costs and utilization of treatment in patients with hemophilia.
Observational study in BMC health services research, 2015. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 21 papers, 1 of them a synthesis that pooled it.
What it found
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
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.
Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.
Who cites it
21 citing papers in PubMed, 1 synthesis or guideline pooled it, 58 citations in OpenAlex.
- Burden of mild haemophilia A: Systematic literature review.Haemophilia : the official journal of the World Federation of Hemophilia · 2019Pooled it
- An analysis of attitudes toward gene therapy in people with severe hemophilia in Germany, a survey-based cross-sectional study.Therapeutic advances in hematology · 2026Article
- Lifetime economic burden of hemophilia using a nationwide real-world healthcare data.PloS one · 2025Article
- Estimating the impact of improved management of haemophilia a on clinical outcomes and healthcare utilisation and costs.BMC research notes · 2023Article
- Management of haemophilia A with inhibitors: A regional cross-talk.Haemophilia : the official journal of the World Federation of Hemophilia · 2022Review
- Health care costs and resource utilization among commercially insured adult patients with hemophilia A managed with FVIII prophylaxis in the United States.Journal of managed care & specialty pharmacy · 2022Article
- Medical costs and hospital utilization for hemophilia A and B urban inpatients in China: a national cross-sectional study.BMC health services research · 2022Article
- The impact of improving haemophilia A management within the Spanish National Healthcare System: a social return on investment analysis.BMC health services research · 2022Article
- Health care resource utilization and costs among adult patients with hemophilia A on factor VIII prophylaxis: an administrative claims analysis.Journal of managed care & specialty pharmacy · 2021Article
- Long-Term Follow-Up of a Portuguese Single-Centre Cohort of Persons with Haemophilia and Hepatitis C Virus Infection.GE Portuguese journal of gastroenterology · 2021Article
- Experience of Immune Tolerance Induction Therapy for Hemophilia A Patients with Inhibitors from a Single Center in India.Indian journal of hematology & blood transfusion : an official journal of Indian Society of Hematology and Blood Transfusion · 2020Review
- Medical expenditure for patients with hemophilia in urban China: data from medical insurance information system from 2013 to 2015.Orphanet journal of rare diseases · 2020Article
- Cost-Effectiveness Analysis of Recombinant Factor VIII Fc-Fusion Protein (rFVIIIFc) for the Treatment of Severe Hemophilia A in Italy Incorporating Real-World Dosing and Joint Health Data.PharmacoEconomics - open · 2020Article
- Haemophilia A: health and economic burden of a rare disease in Portugal.Orphanet journal of rare diseases · 2019Article
- How to discuss gene therapy for haemophilia? A patient and physician perspective.Haemophilia : the official journal of the World Federation of Hemophilia · 2019Review
- Budget impact analysis of the use of extended half-life recombinant factor VIII (efmoroctocog alfa) for the treatment of congenital haemophilia a: the Italian National Health System perspective.BMC health services research · 2018Article
- Value co-creation in healthcare: evidence from innovative therapeutic alternatives for hereditary angioedema.BMC health services research · 2018Article
- Modern Treatments of Haemophilia: Review of Cost-Effectiveness Analyses and Future Directions.PharmacoEconomics · 2018Review
- An investigational RNAi therapeutic targeting antithrombin for the treatment of hemophilia A and B.Journal of blood medicine · 2018Review
- The cost of severe haemophilia in Europe: the CHESS study.Orphanet journal of rare diseases · 2017Article
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
4 authors at 2 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
backgroundAlthough hemophilia has a potentially high economic impact, there are no published estimates of healthcare costs for this disease in Portugal. The aim of this study was to evaluate costs of treatment and hospital utilization among patients with hemophilia A and B, with and without inhibitors, over a 3-year period in a Portuguese Comprehensive Care Hemophilia Centre. This is the first study on the financial impact of healthcare costs in patients with hemophilia in Portugal.
methodsThis retrospective, observational study identified patients diagnosed with hemophilia A and B using medical and pharmacy electronic medical records and data from Centro Hospitalar São João, between January 2011 and December 2013. Patients with inhibitors were all high responders (>5 Bethesda Units [BU]). Severity was classified as mild, moderate or severe based on clotting factor levels. Two main outcomes were measured: (1) cost associated with hospital pharmacy claims (clotting factor) and (2) number of hospital visits/hospitalization.
resultsA cohort of 103 patients were identified: 72 (69.9 %) with hemophilia A and 31 (30.1 %) with hemophilia B. Among these, five individuals were classified as patients with inhibitors (four with hemophilia A and one with hemophilia B). From the cohort of hemophilia A patients, 36 individuals (35.0 %) were identified as having severe disease; 20 (19.4 %) moderate; and 16 (15.5 %) mild. In the cohort of hemophilia B patients, 14 (13.6 %) were identified as having severe disease; 14 (13.6 %) moderate; and three (2.9 %) mild. The total mean aggregate cost per year (including clotting factor and hospital utilization) for patients with severe hemophilia B was €112,469, compared with €793 for mild hemophilia A. Clotting factor concentrate amounted for 90 % of total cost in severe cases and hospital utilization was also higher in these cases.
conclusionsHemophilia treatment is expensive, particularly for patients with severe disease and especially if they develop inhibitors to replacement clotting factors. In our study, severe hemophilia is associated with greater annual total costs in both types of hemophilia (A = €77,587 and B = € 112,469). Patients with inhibitors have costs 3.3 times higher than patients without inhibitors. Age was not associated with significantly greater total costs (clotting factor and hospital visits/hospitalizations).
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