Marianna Cavazza1, Yllka Kodra2, Patrizio Armeni3, Marta De Santis2, Julio López-Bastida4,5, Renata Linertová5,6, Juan Oliva-Moreno5,7, Pedro Serrano-Aguilar5,8, Manuel Posada-de-la-Paz9, Domenica Taruscio2, Arrigo Schieppati10, Georgi Iskrov11,12, László Gulácsi13, Johann Matthias Graf von der Schulenburg14, Panos Kanavos15, Karine Chevreul16,17,18, Ulf Persson19, Giovanni Fattore3. 1. Centre for Research on Health and Social Care Management (CERGAS), Bocconi University, Via Roentgen 1, 20136, Milan, Italy. marianna.cavazza@unibocconi.it. 2. National Centre for Rare Diseases, Istituto Superiore di Sanità (ISS), Rome, Italy. 3. Centre for Research on Health and Social Care Management (CERGAS), Bocconi University, Via Roentgen 1, 20136, Milan, Italy. 4. University of Castilla-La Mancha, Talavera de la Reina, Toledo, Spain. 5. Red de Investigación en Servicios Sanitarios en Enfermedades Crónicas (REDISSEC), Madrid, Spain. 6. Fundación Canaria de Investigación Sanitaria (FUNCANIS), Las Palmas de Gran Canaria, Spain. 7. University of Castilla-La Mancha, Toledo, Spain. 8. Evaluation and Planning Service at Canary Islands Health Service, Santa Cruz de Tenerife, Spain. 9. Institute of Rare Diseases Research, ISCIII, SpainRDR & CIBERER, Madrid, Spain. 10. "Aldo and Cele Daccò" Clinical Research Center for Rare Diseases, Mario Negri Institute for Pharmacological Research, Ranica (Bergamo), Italy. 11. Institute of Rare Diseases, Plovdiv, Bulgaria. 12. Department of Social Medicine and Public Health, Faculty of Public Health, Medical University of Plovdiv, Plovdiv, Bulgaria. 13. Department of Health Economics, Corvinus University of Budapest, Budapest, Hungary. 14. Centre for Health Economics Research Hannover (CHERH), Leibniz Universität Hannover, Hanover, Germany. 15. Department of Social Policy and LSE Health, London School of Economics and Political Science, London, UK. 16. URC Eco Ile de France, AP-HP, Paris, France. 17. Université Paris Diderot, Sorbonne Paris Cité, ECEVE, UMRS 1123, Paris, France. 18. INSERM, ECEVE, U1123, Paris, France. 19. The Swedish Institute for Health Economics, Lund, Sweden.
Abstract
OBJECTIVE: The 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. METHODS: We 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. RESULTS: A 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. CONCLUSION: We 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.
OBJECTIVE: The 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. METHODS: We 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. RESULTS: A 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. CONCLUSION: We 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.
Entities:
Keywords:
Haemophilia; Health related quality of life; Rare diseases; Social burden; Societal costs
Authors: A Tagliaferri; G F Rivolta; A Iorio; E Oliovecchio; M E Mancuso; M Morfini; A Rocino; M G Mazzucconi; M Franchini; N Ciavarella; A Scaraggi; L Valdrè; G Tagariello; P Radossi; G Muleo; P G Iannaccaro; C Biasoli; D Vincenzi; M L Serino; S Linari; C Molinari; E Boeri; M La Pecorella; M T Carloni; E Santagostino; G Di Minno; A Coppola; A Rocino; E Zanon; L Spiezia; C Di Perna; M Marchesini; M Marcucci; A Dragani; S Macchi; P Albertini; M D'Incà; C Santoro; F Biondo; G Piseddu; G Rossetti; G Barillari; G Gandini; A C Giuffrida; G Castaman Journal: Haemophilia Date: 2010-02-09 Impact factor: 4.287
Authors: K Steen Carlsson; S Höjgård; A Glomstein; S Lethagen; S Schulman; L Tengborn; A Lindgren; E Berntorp; B Lindgren Journal: Haemophilia Date: 2003-09 Impact factor: 4.287