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

authored by

BURQOL-RD Research Network , Karine Chevreul, Coralie Gandré, Karen Berg Brigham, Julio López-Bastida, Renata Linertová, Juan Oliva-Moreno, Pedro Serrano-Aguilar, Manuel Posada-de-la-Paz, Domenica Taruscio, Arrigo Schieppati, Georgi Iskrov, László Gulácsi, Johann Matthias Graf von der Schulenburg, Panos Kanavos, Ulf Persson, Giovani Fattore

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.

Organisation(s)

Center for Health Economics Research Hannover (CHERH)

External Organisation(s)

AP-HP Assistance Publique - Hopitaux de Paris
Institut national de la santé et de la recherche médicale (INSERM)
Universite Paris XII
Universidad de Castilla-La Mancha
Red de Investigación en Servicios Sanitarios en Enfermedades Crónicas (REDISSEC)
Canary Islands Foundation for Health Research (FUNCANIS)
Istituto Superiore di Sanita
Institute of Pharmacological Research Mario Negri IRCCS
Institute of Rare Diseases
Medical University of Plovdiv
Corvinus University of Budapest
London School of Economics and Political Science
The Swedish Institute for Health Economics
Università Commerciale Luigi Bocconi
Universite Paris 7
Canary Islands Health Service
Institute of Health Carlos III (ISCIII)

Type

Article

Journal

European Journal of Health Economics

Volume

17

Pages

43-52

No. of pages

10

ISSN

1618-7598

Publication date

04.2016

Publication status

Published

Peer reviewed

Yes

ASJC Scopus subject areas

Economics, Econometrics and Finance (miscellaneous), Health Policy

Sustainable Development Goals

SDG 3 - Good Health and Well-being

Electronic version(s)

https://doi.org/10.1007/s10198-016-0784-3 (Access: Closed)