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Social/economic costs and health-related quality of life in patients with scleroderma in Europe

Author

Listed:
  • Julio López-Bastida

    (Universidad de Castilla-La Mancha
    Red de Investigación en Servicios Sanitarios en Enfermedades Crónicas (REDISSEC))

  • Renata Linertová

    (Red de Investigación en Servicios Sanitarios en Enfermedades Crónicas (REDISSEC)
    Fundación Canaria de Investigación Sanitaria (FUNCANIS))

  • Juan Oliva-Moreno

    (Red de Investigación en Servicios Sanitarios en Enfermedades Crónicas (REDISSEC)
    Universidad de Castilla-La Mancha)

  • Pedro Serrano-Aguilar

    (Red de Investigación en Servicios Sanitarios en Enfermedades Crónicas (REDISSEC)
    Evaluation and Planning Service at Canary Islands Health Service)

  • Manuel Posada-de-la-Paz

    (SpainRDR and CIBERER)

  • Panos Kanavos

    (London School of Economics and Political Science)

  • Domenica Taruscio

    (Istituto Superiore di Sanità (ISS))

  • Arrigo Schieppati

    (Istituto di Ricerche Farmacologiche Mario Negri)

  • Georgi Iskrov

    (Institute of Rare Diseases
    Medical University of Plovdiv)

  • Márta Péntek

    (Corvinus University of Budapest)

  • Claudia Delgado

    (Federación Española de Enfermedades Raras (FEDER))

  • Johann Mathias Schulenburg

    (Leibniz Universität Hannover)

  • Ulf Persson

    (Swedish Institute for Health Economics)

  • Karine Chevreul

    (URC Eco Ile de France, AP-HP
    Sorbonne Paris Cité, ECEVE, UMRS 1123
    INSERM, ECEVE, U1123)

  • Giovanni Fattore

    (Bocconi University)

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 systemic sclerosis (SSc; scleroderma) in Europe. Methods We conducted a cross-sectional study of patients with SSc (involving both localised and systemic sclerosis) from Germany, Italy, Spain, France, the UK, Hungary and Sweden. Data on demographic characteristics, healthcare resource utilisation, informal care, labour 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) questionnaire. Results A total of 589 patients completed the questionnaire. The rate of patients with localised scleroderma, limited cutan and diffuse cutan SSc were 28, 68 and 4 %, respectively. Average annual costs varied from country to country and ranged from € 4607 to € 30,797 (reference year: 2012). Estimated direct healthcare costs ranged from € 1413 to € 17,300; direct non-healthcare costs ranged from € 1875 to € 4684 and labour productivity losses ranged from € 1701 to € 14,444. The mean EQ-5D index score for adult SSc patients varied from 0.49 to 0.75 and the mean EQ-5D visual analogue scale score was between 58.72 and 65.86. Conclusion The main strengths of this study lie in our bottom-up approach to costing and our evaluation of SSs patients from a broad societal perspective. This type of analysis is very unusual in the international literature on rare diseases in comparison with other illnesses. We concluded that SSc patients incur considerable societal costs and experience substantial deterioration in HRQOL.

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  • Julio López-Bastida & Renata Linertová & Juan Oliva-Moreno & Pedro Serrano-Aguilar & Manuel Posada-de-la-Paz & Panos Kanavos & Domenica Taruscio & Arrigo Schieppati & Georgi Iskrov & Márta Péntek & Cl, 2016. "Social/economic costs and health-related quality of life in patients with scleroderma in Europe," The European Journal of Health Economics, Springer;Deutsche Gesellschaft für Gesundheitsökonomie (DGGÖ), vol. 17(1), pages 109-117, April.
    • Handle: RePEc:spr:eujhec:v:17:y:2016:i:1:d:10.1007_s10198-016-0789-y
    DOI: 10.1007/s10198-016-0789-y
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    1. Linertová, Renata & Serrano-Aguilar, Pedro & Posada-de-la-Paz, Manuel & Hens-Pérez, Manuel & Kanavos, Panos & Taruscio, Domenica & Schieppati, Arrigo & Stefanov, Rumen & Péntek, Márta & Delgado, Claud, 2012. "Delphi approach to select rare diseases for a European representative survey. The BURQOL-RD study," Health Policy, Elsevier, vol. 108(1), pages 19-26.
    2. Angelis, Aris & Tordrup, David & Kanavos, Panos, 2015. "Socio-economic burden of rare diseases: A systematic review of cost of illness evidence," Health Policy, Elsevier, vol. 119(7), pages 964-979.
    3. Bernard Berg & Werner Brouwer & Marc Koopmanschap, 2004. "Economic valuation of informal care," The European Journal of Health Economics, Springer;Deutsche Gesellschaft für Gesundheitsökonomie (DGGÖ), vol. 5(1), pages 36-45, February.
    4. Karine Chevreul & Morgane Michel & Karen Berg Brigham & Julio López-Bastida & Renata Linertová & Juan Oliva-Moreno & Pedro Serrano-Aguilar & Manuel Posada-de-la-Paz & Domenica Taruscio & Arrigo Schiep, 2016. "Social/economic costs and health-related quality of life in patients with cystic fibrosis in Europe," The European Journal of Health Economics, Springer;Deutsche Gesellschaft für Gesundheitsökonomie (DGGÖ), vol. 17(1), pages 7-18, April.
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