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

Methods

We conducted a cross-sectional study of patients with histiocytosis from France, Germany, Italy, Spain, Bulgaria, the UK, and Sweden. Data on demographic characteristics, health resource utilisation, informal care, loss of labour productivity 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 134 patients (35 France, 32 Germany, 30 Italy, 24 Spain, 7 Bulgaria, 4 UK and 2 Sweden) completed the questionnaire. The average annual costs ranged from € 6832 to € 33,283 between countries, the year of reference being 2012. Estimated direct healthcare costs ranged from € 1698 to € 18,213; direct nonhealthcare costs ranged from € 2936 to € 17,622 and labour productivity losses ranged from € 1 to € 8855. The mean EQ-5D score for adult histiocytosis patients was estimated at between 0.32 and 0.85, and the mean EQ-5D visual analogue scale score was estimated at between 50.00 and 66.50.

Conclusion

The main strengths of this study lie in our bottom-up approach to costing and in the evaluation of histiocytosis patients from a broad perspective (societal costs). This type of analysis is very scarce in international literature for rare diseases in comparison with other illnesses. We conclude that histiocytosis patients incur considerable societal costs and experience substantial deterioration in HRQOL.

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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; Consulta Nazionale delle Malattie Rare, Italy; AIRI LCH Onlus - Associazione Italiana Ricerca Istiocitos, Italyi; Rare Diseases Sweden; Federación Española de Efermedades Raras (FEDER), Spain; Rare Disease UK; Euro-Histio-Net and Rare Diseases Europe (EURORDIS); Euro-Histio-Net; Association Histiocytose France; Histiozytosehilfe e.V., Germany; AIRI LCH Onlus– Associazione Italiana Ricerca Istiocitosi, Italy; Föräldraföreningen för barn med Histiocytos, Sweden; Asociación Española contra la Histiocitosis, Spain.

Author information

Authors and Affiliations

  1. Institute for Rare Diseases, 22 Maestro G. Atanasov Street, 4023, Plovdiv, Bulgaria

    Georgi Iskrov & Rumen Stefanov

  2. Department of Pediatrics, BioCruces Health Research Institute, Hospital Universitario Cruces, Bizkaia, Spain

    Itziar Astigarraga

  3. University of Castilla-La Mancha, Talavera de la Reina, Toledo, Spain

    Julio López-Bastida

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

    Julio López-Bastida, Renata Linertová, Juan Oliva-Moreno & Pedro Serrano-Aguilar

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

    Renata Linertová

  6. University of Castilla-La Mancha, Toledo, Spain

    Juan Oliva-Moreno

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

    Pedro Serrano-Aguilar

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

    Manuel Posada-de-la-Paz

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

    Arrigo Schieppati

  10. National Centre for Rare Diseases, Istituto Superiore di Sanità (ISS), Rome, Italy

    Domenica Taruscio

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

    Márta Péntek

  12. Centre for Health Economics Research Hannover (CHERH), Leibniz Universität Hannover, Hannover, Germany

    Johann Matthias Graf von der Schulenburg

  13. Department of Social Policy and LSE Health, London School of Economics and Political Science, London, UK

    Panos Kanavos

  14. URC Eco Ile de France, AP-HP, Hôtel Dieu, Paris, France

    Karine Chevreul

  15. The Swedish Institute for Health Economics, Lund, Sweden

    Ulf Persson

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

    Giovanni Fattore

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

    Georgi Iskrov & Rumen Stefanov

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

    Karine Chevreul

  19. INSERM, ECEVE, U1123, Paris, France

    Karine Chevreul

Authors
  1. Georgi Iskrov
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  2. Itziar Astigarraga
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  3. Rumen Stefanov
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  4. Julio López-Bastida
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  5. Renata Linertová
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  7. Pedro Serrano-Aguilar
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  8. Manuel Posada-de-la-Paz
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  9. Arrigo Schieppati
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  10. Domenica Taruscio
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  11. Márta Péntek
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  12. Johann Matthias Graf von der Schulenburg
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  13. Panos Kanavos
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  14. Karine Chevreul
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  15. Ulf Persson
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  16. Giovanni Fattore
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Consortia

BURQOL-RD Research Network

Corresponding author

Correspondence to Georgi Iskrov.

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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 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.

Conflict of interest

The authors declare that they have no conflicts of interest.

Additional information

Members of the BURQOL-RD Research Network listed in Appendix 1 of the ESM.

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Iskrov, G., Astigarraga, I., Stefanov, R. et al. Social/economic costs and health-related quality of life in patients with histiocytosis in Europe. Eur J Health Econ 17 (Suppl 1), 67–78 (2016). https://doi.org/10.1007/s10198-016-0790-5

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  • DOI: https://doi.org/10.1007/s10198-016-0790-5

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