The economic burden and health-related quality of life associated with systemic sclerosis in France

Abstract

Objectives: To provide data on the economic burden and health-related quality of life (HRQoL) associated with systemic sclerosis (SSc) in France and to raise awareness of the repercussions of this disease for patients and caregivers and on the health and social care system.

Method: A cross-sectional study was carried out on 147 patients recruited through the Association des Sclérodermiques de France (ASF), the French association for SSc patients. Data on the patients’ use of resources were obtained retrospectively from an online questionnaire and costs were estimated by a bottom-up approach. The HRQoL patients and caregivers was assessed with the five-level EURQol-5 Dimension (EQ-5D-5L) health questionnaire.

Results: The average annual cost of SSc was estimated at EUR 22 459 per patient. Direct healthcare costs amounted to EUR 8452, direct non-healthcare formal costs to EUR 1606, direct non-healthcare informal costs to EUR 1875, and indirect costs resulting from patients’ absence from the labour market to EUR 10 526. The main contributors to SSc costs were hospitalizations and early retirement. Mean EQ-5D utility scores were 0.49 for patients and 0.66 for caregivers.

Conclusions: Although SSc is a rare disease, its economic burden from a societal perspective is substantial and the consequences for HRQoL are significant for both patients and caregivers in France, underscoring the need to develop tailored policies targeted at improving patients’ care and reducing the long-term impact of SSc.

Acknowledgements

We are grateful for the strong support of Dominique Godard from ASF in organizing patient recruitment. We also thank Alexia Poupard of the Alliance Maladies Rares, our collaborating partner in BURQOL-RD, for her very important role in coordinating this survey. We also gratefully acknowledge Julien Thillard for his statistical support, Amélie Prigent for her valuable comments, and two anonymous reviewers for their helpful suggestions.

This study was 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 in 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 here.

Appendix I: BURQOL-RD Research Network

Canary Islands Foundation for Research and Health (FUNCIS), Spain: Pedro Serrano-Aguilar, Renata Linertová. University of Castilla-La Mancha, Spain: Julio López-Bastida, Juan Oliva-Moreno. Research Institute for Rare Diseases, Institute of Health Carlos III, Spain: Manuel Posada de la Paz, Manuel Hens Pérez, Ignacio Abaitua. National Center for Rare Diseases, the National Institute of Health, Italy: Domenica Taruscio, Yllka Kodra. Mario Negri Institute for Pharmacological Research, Italy: Arrigo Schieppati. Bulgarian Association for Promotion of Education and Science, Bulgaria: Rumen Stefanov, Georgi Iskrov. Centre for Public Affairs Studies Foundation,Hungary: László Gulácsi, Márta Péntek. The Spanish Federation for Rare Diseases, Spain: Rosa Sánchez de Vega García, Claudia Delgado. London School of Economics and Political Science, United Kingdom: Panos Kanavos, Aris Angelis, Elena Nicod. Leibniz University Hannover, Germany: Johann-Matthias Graf von der Schulenburg, Alexander Kuhlmann. The Swedish Institute for Health Economics, Sweden: Ulf Persson, Ola Ghatnekar. University of Paris Est, Franc): Karine Chevreul, Karen Brigham. University of Business, Luigi Bocconi, Italy: Giovanni Fattore, Marianna Cavazza.