Social/economic costs and health-related quality of life in patients with fragile X syndrome in Europe

Karine Chevreul (), Coralie Gandré, Karen Berg Brigham, Julio López-Bastida, Renata Linertová, Juan Oliva (), Pedro Serrano-Aguilar, Manuel Posada- de-la-Paz, Domenica Taruscio, Arrigo Schieppati, Georgi Iskrov, László Gulácsi, Johann Matthias Graf Schulenburg, Panos Kanavos, Ulf Persson and Giovani Fattore
Additional contact information
Karine Chevreul: URC Eco Ile de France, AP-HP
Coralie Gandré: URC Eco Ile de France, AP-HP
Karen Berg Brigham: URC Eco Ile de France, AP-HP
Julio López-Bastida: University of Castilla-La Mancha
Renata Linertová: Red de Investigación en Servicios Sanitarios en Enfermedades Crónicas (REDISSEC)
Pedro Serrano-Aguilar: Red de Investigación en Servicios Sanitarios en Enfermedades Crónicas (REDISSEC)
Manuel Posada- de-la-Paz: Institute of Rare Diseases Research, ISCIII, SpainRDR & CIBERER
Domenica Taruscio: National Center for Rare Diseases, Istituto Superiore di Sanità (ISS)
Arrigo Schieppati: Istituto di Ricerche Farmacologiche Mario Negri
Georgi Iskrov: Institute of Rare Diseases
László Gulácsi: Corvinus University of Budapest
Johann Matthias Graf Schulenburg: Leibniz Universität Hannover
Panos Kanavos: London School of Economics and Political Science
Ulf Persson: The Swedish Institute for Health Economics
Giovani Fattore: Bocconi University

The European Journal of Health Economics, 2016, vol. 17, issue 1, No 5, 43-52

Abstract: Abstract Objective To estimate the social/economic costs of fragile X syndrome (FXS) in Europe and to assess the health-related quality of life (HRQOL) of patients and caregivers. Methods A cross-sectional study was conducted in a sample of European countries. Patients were recruited through patients’ associations. Data on their resource use and absence from the labour market were retrospectively obtained from an online questionnaire. Costs were estimated by a bottom-up approach and the EuroQol-5 Domain (EQ-5D) questionnaire was used to measure patients’ and caregivers’ HRQOL. Results Five countries were included in the analysis. The mean annual cost of FXS per patient varied from €4951 in Hungary to €58,862 in Sweden. Direct non-healthcare costs represented the majority of costs in all countries but there were differences in the share incurred by formal and informal care among those costs. Costs were also shown to differ between children and adults. Mean EQ-5D utility score for adult patients varied from 0.52 in France (n = 42) to 0.73 in Hungary (n = 2), while for caregivers this score was consistently inferior to 0.87. Conclusion Our findings underline that, although its prevalence is low, FXS is costly from a societal perspective. They support the development of tailored policies to reduce the consequences of FXS on both patients and their relatives.

Keywords: Fragile X syndrome; Economic burden; Cost analysis; Quality of life (search for similar items in EconPapers)
Date: 2016
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Citations: View citations in EconPapers (16)

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DOI: 10.1007/s10198-016-0784-3

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