Georgi Iskrov1,2, Itziar Astigarraga3, Rumen Stefanov4,5, Julio López-Bastida6,7, Renata Linertová7,8, Juan Oliva-Moreno7,9, Pedro Serrano-Aguilar7,10, Manuel Posada-de-la-Paz11, Arrigo Schieppati12, Domenica Taruscio13, Márta Péntek14, Johann Matthias Graf von der Schulenburg15, Panos Kanavos16, Karine Chevreul17,18,19, Ulf Persson20, Giovanni Fattore21. 1. Institute for Rare Diseases, 22 Maestro G. Atanasov Street, 4023, Plovdiv, Bulgaria. iskrov@raredis.org. 2. Department of Social Medicine and Public Health, Faculty of Public Health, Medical University of Plovdiv, Plovdiv, Bulgaria. iskrov@raredis.org. 3. Department of Pediatrics, BioCruces Health Research Institute, Hospital Universitario Cruces, Bizkaia, Spain. 4. Institute for Rare Diseases, 22 Maestro G. Atanasov Street, 4023, Plovdiv, Bulgaria. 5. Department of Social Medicine and Public Health, Faculty of Public Health, Medical University of Plovdiv, Plovdiv, Bulgaria. 6. University of Castilla-La Mancha, Talavera de la Reina, Toledo, Spain. 7. Red de Investigación en Servicios Sanitarios en Enfermedades Crónicas (REDISSEC), Madrid, Spain. 8. Fundación Canaria de Investigación Sanitaria (FUNCANIS), Las Palmas de Gran Canaria, Spain. 9. University of Castilla-La Mancha, Toledo, Spain. 10. Evaluation and Planning Service at Canary Islands Health Service, Santa Cruz de Tenerife, Spain. 11. Institute of Rare Diseases Research, ISCIII, SpainRDR & CIBERER, Madrid, Spain. 12. Centro di Ricerche Cliniche per Malattie Rare Aldo e Cele Daccò, Istituto di Ricerche Farmacologiche Mario Negri, Ranica, Bergamo, Italy. 13. National Centre for Rare Diseases, Istituto Superiore di Sanità (ISS), Rome, Italy. 14. Department of Health Economics, Corvinus University of Budapest, Budapest, Hungary. 15. Centre for Health Economics Research Hannover (CHERH), Leibniz Universität Hannover, Hannover, Germany. 16. Department of Social Policy and LSE Health, London School of Economics and Political Science, London, UK. 17. URC Eco Ile de France, AP-HP, Hôtel Dieu, Paris, France. 18. Université Paris Diderot, Sorbonne Paris Cité, ECEVE, UMRS 1123, Paris, France. 19. INSERM, ECEVE, U1123, Paris, France. 20. The Swedish Institute for Health Economics, Lund, Sweden. 21. Centre for Research on Health and Social Care Management (CERGAS), Bocconi University, Milan, Italy.
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 histiocytosis in Europe. METHODS: We conducted a cross-sectional study of patients with histiocytosis from France, Germany, Italy, Spain, Bulgaria, the UK, and Sweden. 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. RESULTS: A total of 134 patients (35 France, 32 Germany, 30 Italy, 24 Spain, 7 Bulgaria, 4 UK and 2 Sweden) completed the questionnaire. The average annual costs ranged from € 6832 to € 33,283 between countries, the year of reference being 2012. Estimated direct healthcare costs ranged from € 1698 to € 18,213; direct nonhealthcare costs ranged from € 2936 to € 17,622 and labour productivity losses ranged from € 1 to € 8855. The mean EQ-5D score for adult histiocytosis patients was estimated at between 0.32 and 0.85, and the mean EQ-5D visual analogue scale score was estimated at between 50.00 and 66.50. CONCLUSION: The main strengths of this study lie in our bottom-up approach to costing and in the evaluation of histiocytosis patients from a broad perspective (societal costs). This type of analysis is very scarce in international literature for rare diseases in comparison with other illnesses. We conclude that histiocytosis patients incur considerable societal costs and experience substantial deterioration in HRQOL.
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 histiocytosis in Europe. METHODS: We conducted a cross-sectional study of patients with histiocytosis from France, Germany, Italy, Spain, Bulgaria, the UK, and Sweden. 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. RESULTS: A total of 134 patients (35 France, 32 Germany, 30 Italy, 24 Spain, 7 Bulgaria, 4 UK and 2 Sweden) completed the questionnaire. The average annual costs ranged from € 6832 to € 33,283 between countries, the year of reference being 2012. Estimated direct healthcare costs ranged from € 1698 to € 18,213; direct nonhealthcare costs ranged from € 2936 to € 17,622 and labour productivity losses ranged from € 1 to € 8855. The mean EQ-5D score for adult histiocytosis patients was estimated at between 0.32 and 0.85, and the mean EQ-5D visual analogue scale score was estimated at between 50.00 and 66.50. CONCLUSION: The main strengths of this study lie in our bottom-up approach to costing and in the evaluation of histiocytosis patients from a broad perspective (societal costs). This type of analysis is very scarce in international literature for rare diseases in comparison with other illnesses. We conclude that histiocytosis patients incur considerable societal costs and experience substantial deterioration in HRQOL.
Entities:
Keywords:
Cost-of-illness; Europe; Health-related quality of life; Histiocytosis; Social cost
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