ArticleThe European journal of health economics : HEPAC : health economics in prevention and care2016
Social/economic costs and health-related quality of life in patients with fragile X syndrome in Europe.
Article in The European journal of health economics : HEPAC : health economics in prevention and care, 2016. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 21 papers, 3 of them syntheses that pooled it.
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Who cites it
21 citing papers in PubMed, 3 syntheses or guidelines pooled it, 43 citations in OpenAlex.
- Impact of Choice of Tariff When Calculating Clinically Meaningful EQ-5D Scores in Metabolic Dysfunction-Associated Steatohepatitis.Liver international : official journal of the International Association for the Study of the Liver · 2025Pooled it
- Cost-of-illness studies in nine Central and Eastern European countries.The European journal of health economics : HEPAC : health economics in prevention and care · 2019Pooled it
- Spillover Effects on Caregivers' and Family Members' Utility: A Systematic Review of the Literature.PharmacoEconomics · 2019Pooled it
- Health-related quality of life and medication use among individuals with Angelman syndrome.Quality of life research : an international journal of quality of life aspects of treatment, care and rehabilitation · 2023Trial
- The Healthcare and Societal Costs of Familial Intellectual Disability.International journal of environmental research and public health · 2024Article
- Methods used to account for caregivers' sex and gender within studies examining the financial burden of caregivers of children and adolescents : Results from a scoping review.ClinicoEconomics and outcomes research : CEOR · 2024Article
- Health-related quality of life in patients with diverse rare diseases: An online survey.Genetics in medicine open · 2024Article
- Are We Capturing the Socioeconomic Burden of Rare Genetic Disease? A Scoping Review of Economic Evaluations and Cost-of-Illness Studies.PharmacoEconomics · 2023Article
- Rare disease emerging as a global public health priority.Frontiers in public health · 2022Review
- Valuing the "Burden" and Impact of Rare Diseases: A Scoping Review.Frontiers in pharmacology · 2022Article
- Cost-of-illness studies in rare diseases: a scoping review.Orphanet journal of rare diseases · 2021Article
- Reflections on the Importance of Cost of Illness Analysis in Rare Diseases: A Proposal.International journal of environmental research and public health · 2021Article
- The Economic Impact and Health-Related Quality of Life of Spinal Muscular Atrophy. An Analysis across Europe.International journal of environmental research and public health · 2020Article
- Generic preference-based health-related quality of life in children with neurodevelopmental disorders: a scoping review.Developmental medicine and child neurology · 2020Article
- Pharmacologic Interventions for Irritability, Aggression, Agitation and Self-Injurious Behavior in Fragile X Syndrome: An Initial Cross-Sectional Analysis.Journal of autism and developmental disorders · 2019Article
- An Electronic Questionnaire for Liver Assessment in Congenital Disorders of Glycosylation (LeQCDG): A Patient-Centered Study.JIMD reports · 2019Article
- Patient preferences in Italy: health state utilities associated with attributes of weekly injection devices for treatment of type 2 diabetes.Patient preference and adherence · 2018Article
- The feasibility of using the EQ-5D-3L with adults with mild to moderate learning disabilities within a randomized control trial: a qualitative evaluation.Pilot and feasibility studies · 2018Article
- Social/economic costs and health-related quality of life in patients with spinal muscular atrophy (SMA) in Spain.Orphanet journal of rare diseases · 2017Article
- Fragile X syndrome: a review of clinical and molecular diagnoses.Italian journal of pediatrics · 2017Review
Corrections and comments
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Authors and funding
17 authors at 14 institutions in 8 countries.
Funding
No grant is acknowledged in the PubMed record.
Abstract
objectiveTo 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.
methodsA 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.
resultsFive 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.
conclusionOur 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.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.