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Social/economic costs and health-related quality of life in patients with juvenile idiopathic arthritis in Europe

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Abstract

Objective

The aim of this study was to determine the economic burden from a societal perspective and the health-related quality of life (HRQOL) of patients with juvenile idiopathic arthritis (JIA) in Europe.

Methods

We conducted a cross-sectional study of patients with JIA from Germany, Italy, Spain, France, the United Kingdom, Bulgaria, and Sweden. Data on demographic characteristics, healthcare resource utilization, informal care, labor productivity losses, and HRQOL were collected from the questionnaires completed by patients or their caregivers. HRQOL was measured with the EuroQol 5-domain (EQ-5D-5L) questionnaire.

Results

A total of 162 patients (67 Germany, 34 Sweden, 33 Italy, 23 United Kingdom, 4 France, and 1 Bulgaria) completed the questionnaire. Excluding Bulgarian results, due to small sample size, country-specific annual health care costs ranged from €18,913 to €36,396 (reference year: 2012). Estimated direct healthcare costs ranged from €11,068 to €22,138; direct non-healthcare costs ranged from €7837 to €14,155 and labor productivity losses ranged from €0 to €8715. Costs are also shown to differ between children and adults. The mean EQ-5D index score for JIA patients was estimated at between 0.44 and 0.88, and the mean EQ-5D visual analogue scale score was estimated at between 62 and 79.

Conclusions

JIA patients incur considerable societal costs and experience substantial deterioration in HRQOL in some countries. Compared with previous studies, our results show a remarkable increase in annual healthcare costs for JIA patients. Reasons for the increase are the inclusion of non-professional caregiver costs, a wider use of biologics, and longer hospital stays.

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Acknowledgments

The authors wish to thank: National Alliance of People with Rare Diseases (NAPRD), Bulgaria; Alliance Maladies Rares, France; ACHSE, Germany; Hungarian Federation of People with Rare and Congenital Diseases (RIROSZ), Hungary; Federazione Italiana Malattie Rare (UNIAMO), Italy; the Consulta Nazionale delle Malattie Rare, Italy; Rare Diseases Sweden; Federación Española de Efermedades Raras (FEDER), Spain; Rare Disease UK and Rare Diseases Europe (EURORDIS); Deutsche Rheuma-Liga Bundesverband e.V., Germany; A.M.R.I Associazione per le malattie reumatiche infantile, Italy; Unga Reumatiker and Reumatikerförbundet, Sweden; NRAS—National Rheumatoid Arthritis Society and Rheumatism Association, UK.

Author information

Authors and Affiliations

  1. Center for Health Economics Research Hannover (CHERH), Leibniz Universität Hannover, Otto-Brenner-Straße 1, 30159, Hannover, Germany

    A. Kuhlmann, T. Schmidt, M. Treskova & J. M. von der Schulenburg

  2. Universidad de Castilla-La Mancha, Talavera de la Reina, Toledo, Spain

    J. López-Bastida

  3. Red de Investigación en Servicios Sanitarios en Enfermedades Crónicas (REDISSEC), Madrid, Spain

    J. López-Bastida, R. Linertová, J. Oliva-Moreno & P. Serrano-Aguilar

  4. Fundación Canaria de Investigación Sanitaria (FUNCANIS), Las Palmas de Gran Canaria, Spain

    R. Linertová

  5. Universidad de Castilla-La Mancha, Toledo, Spain

    J. Oliva-Moreno

  6. Evaluation and Planning Service at Canary Islands Health Service, Santa Cruz de Tenerife, Spain

    P. Serrano-Aguilar

  7. Institute of Rare Diseases Research, ISCIII, SpainRDR & CIBERER, Madrid, Spain

    M. Posada-de-la-Paz

  8. Department of Social Policy and LSE Health, London School of Economics and Political Science, London, United Kingdom

    P. Kanavos

  9. National Center for Rare Diseases, Istituto Superiore di Sanità (ISS), Rome, Italy

    D. Taruscio

  10. Centro di Ricerche Cliniche per Malattie Rare Aldo e Cele Daccò, Istituto di Ricerche Farmacologiche Mario Negri, Ranica (Bergamo), Italy

    A. Schieppati

  11. Institute of Rare Diseases, Plovdiv, Bulgaria

    G. Iskrov

  12. Department of Social Medicine and Public Health, Faculty of Public Health, Medical University of Plovdiv, Plovdiv, Bulgaria

    G. Iskrov

  13. Department of Health Economics, Corvinus University of Budapest, Budapest, Hungary

    M. Péntek

  14. Federación Española de Enfermedades Raras (FEDER), Madrid, Spain

    C. Delgado

  15. Swedish Institute for Health Economics, Lund, Sweden

    U. Persson

  16. URC Eco Ile de France, AP-HP, Paris, France

    K. Chevreul

  17. Université Paris Diderot, Sorbonne Paris Cité, ECEVE, UMRS 1123, Paris, France

    K. Chevreul

  18. INSERM, ECEVE, U1123, Paris, France

    K. Chevreul

  19. Centre for Research on Health and Social Care Management (CERGAS), Bocconi University, Milan, Italy

    G. Fattore

Authors
  1. A. Kuhlmann
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  2. T. Schmidt
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  3. M. Treskova
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  4. J. López-Bastida
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  5. R. Linertová
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  6. J. Oliva-Moreno
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  7. P. Serrano-Aguilar
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  8. M. Posada-de-la-Paz
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  9. P. Kanavos
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  10. D. Taruscio
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  11. A. Schieppati
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  12. G. Iskrov
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  13. M. Péntek
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  14. C. Delgado
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  15. J. M. von der Schulenburg
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  16. U. Persson
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  17. K. Chevreul
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  18. G. Fattore
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Consortia

The BURQOL-RD Research Network

Corresponding author

Correspondence to A. Kuhlmann.

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Funding

Supported by the Social/Economic Burden and Health-Related Quality of Life in Patients with Rare Diseases in Europe Project, which 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.

Conflict of interest

The authors declare that they have no conflicts of interest.

Additional information

Members of the BURQOL-RD Research Network listed in Supplementary Annex 1.

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Kuhlmann, A., Schmidt, T., Treskova, M. et al. Social/economic costs and health-related quality of life in patients with juvenile idiopathic arthritis in Europe. Eur J Health Econ 17 (Suppl 1), 79–87 (2016). https://doi.org/10.1007/s10198-016-0786-1

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  • DOI: https://doi.org/10.1007/s10198-016-0786-1

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