Details
| Original language | English |
|---|---|
| Pages (from-to) | 43-52 |
| Number of pages | 10 |
| Journal | European Journal of Health Economics |
| Volume | 17 |
| Early online date | 12 Apr 2016 |
| Publication status | Published - Apr 2016 |
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
- Cost analysis, Economic burden, Fragile X syndrome, Quality of life
ASJC Scopus subject areas
- Economics, Econometrics and Finance(all)
- Economics, Econometrics and Finance (miscellaneous)
- Medicine(all)
- Health Policy
Sustainable Development Goals
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In: European Journal of Health Economics, Vol. 17, 04.2016, p. 43-52.
Research output: Contribution to journal › Article › Research › peer review
}
TY - JOUR
T1 - Social/economic costs and health-related quality of life in patients with fragile X syndrome in Europe
AU - BURQOL-RD Research Network
AU - Chevreul, Karine
AU - Gandré, Coralie
AU - Brigham, Karen Berg
AU - López-Bastida, Julio
AU - Linertová, Renata
AU - Oliva-Moreno, Juan
AU - Serrano-Aguilar, Pedro
AU - Posada-de-la-Paz, Manuel
AU - Taruscio, Domenica
AU - Schieppati, Arrigo
AU - Iskrov, Georgi
AU - Gulácsi, László
AU - von der Schulenburg, Johann Matthias Graf
AU - Kanavos, Panos
AU - Persson, Ulf
AU - Fattore, Giovani
N1 - Funding Information: Supported by the Social/Economic Burden and Health-Related Quality of Life in Patients with Rare Diseases in Europe Project, which has received funding from the European Union within the framework of the Health Programme (Grant A101205). The Executive Agency of the European Union is not responsible for any use that may be made of the information contained herein.
PY - 2016/4
Y1 - 2016/4
N2 - 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.
AB - 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.
KW - Cost analysis
KW - Economic burden
KW - Fragile X syndrome
KW - Quality of life
UR - http://www.scopus.com/inward/record.url?scp=84963641694&partnerID=8YFLogxK
U2 - 10.1007/s10198-016-0784-3
DO - 10.1007/s10198-016-0784-3
M3 - Article
C2 - 27072054
AN - SCOPUS:84963641694
VL - 17
SP - 43
EP - 52
JO - European Journal of Health Economics
JF - European Journal of Health Economics
SN - 1618-7598
ER -