title: Social/economic costs and health-related quality of life in patients with juvenile idiopathic arthritis in Europe.
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.
authors.affiliation: Center for Health Economics Research Hannover (CHERH), Leibniz Universität Hannover, Otto-Brenner-Straße 1, 30159, Hannover, Germany. ak@cherh.de.
authors.forename: A
authors.initials: A
authors.lastname: Kuhlmann
authors.affiliation: Center for Health Economics Research Hannover (CHERH), Leibniz Universität Hannover, Otto-Brenner-Straße 1, 30159, Hannover, Germany.
authors.forename: T
authors.initials: T
authors.lastname: Schmidt
authors.affiliation: Center for Health Economics Research Hannover (CHERH), Leibniz Universität Hannover, Otto-Brenner-Straße 1, 30159, Hannover, Germany.
authors.forename: M
authors.initials: M
authors.lastname: Treskova
authors.affiliation: Universidad de Castilla-La Mancha, Talavera de la Reina, Toledo, Spain.|Red de Investigación en Servicios Sanitarios en Enfermedades Crónicas (REDISSEC), Madrid, Spain.
authors.forename: J
authors.initials: J
authors.lastname: López-Bastida
authors.affiliation: Red de Investigación en Servicios Sanitarios en Enfermedades Crónicas (REDISSEC), Madrid, Spain.|Fundación Canaria de Investigación Sanitaria (FUNCANIS), Las Palmas de Gran Canaria, Spain.
authors.forename: R
authors.initials: R
authors.lastname: Linertová
authors.affiliation: Red de Investigación en Servicios Sanitarios en Enfermedades Crónicas (REDISSEC), Madrid, Spain.|Universidad de Castilla-La Mancha, Toledo, Spain.
authors.forename: J
authors.initials: J
authors.lastname: Oliva-Moreno
authors.affiliation: Red de Investigación en Servicios Sanitarios en Enfermedades Crónicas (REDISSEC), Madrid, Spain.|Evaluation and Planning Service at Canary Islands Health Service, Santa Cruz de Tenerife, Spain.
authors.forename: P
authors.initials: P
authors.lastname: Serrano-Aguilar
authors.affiliation: Institute of Rare Diseases Research, ISCIII, SpainRDR & CIBERER, Madrid, Spain.
authors.forename: M
authors.initials: M
authors.lastname: Posada-de-la-Paz
authors.affiliation: Department of Social Policy and LSE Health, London School of Economics and Political Science, London, United Kingdom.
authors.forename: P
authors.initials: P
authors.lastname: Kanavos
authors.affiliation: National Center for Rare Diseases, Istituto Superiore di Sanità (ISS), Rome, Italy.
authors.forename: D
authors.initials: D
authors.lastname: Taruscio
authors.affiliation: Centro di Ricerche Cliniche per Malattie Rare Aldo e Cele Daccò, Istituto di Ricerche Farmacologiche Mario Negri, Ranica (Bergamo), Italy.
authors.forename: A
authors.initials: A
authors.lastname: Schieppati
authors.affiliation: Institute of Rare Diseases, Plovdiv, Bulgaria.|Department of Social Medicine and Public Health, Faculty of Public Health, Medical University of Plovdiv, Plovdiv, Bulgaria.
authors.forename: G
authors.initials: G
authors.lastname: Iskrov
authors.affiliation: Department of Health Economics, Corvinus University of Budapest, Budapest, Hungary.
authors.forename: M
authors.initials: M
authors.lastname: Péntek
authors.affiliation: Federación Española de Enfermedades Raras (FEDER), Madrid, Spain.
authors.forename: C
authors.initials: C
authors.lastname: Delgado
authors.affiliation: Center for Health Economics Research Hannover (CHERH), Leibniz Universität Hannover, Otto-Brenner-Straße 1, 30159, Hannover, Germany.
authors.forename: J M
authors.initials: JM
authors.lastname: von der Schulenburg
authors.affiliation: Swedish Institute for Health Economics, Lund, Sweden.
authors.forename: U
authors.initials: U
authors.lastname: Persson
authors.affiliation: URC Eco Ile de France, AP-HP, Paris, France.|Université Paris Diderot, Sorbonne Paris Cité, ECEVE, UMRS 1123, Paris, France.|INSERM, ECEVE, U1123, Paris, France.
authors.forename: K
authors.initials: K
authors.lastname: Chevreul
authors.affiliation: Centre for Research on Health and Social Care Management (CERGAS), Bocconi University, Milan, Italy.
authors.forename: G
authors.initials: G
authors.lastname: Fattore
mesh_terms: D000293:Adolescent; D000328:Adult; D001171:Arthritis, Juvenile / Q000191:economics* / Q000523:psychology; D017028:Caregivers; D002648:Child; D002675:Child, Preschool; D017281:Cost of Illness; D003430:Cross-Sectional Studies; D005060:Europe; D005260:Female; D017048:Health Care Costs / Q000706:statistics & numerical data; D006801:Humans; D008297:Male; D008875:Middle Aged; D005791:Patient Care / Q000191:economics; D011788:Quality of Life; D018582:Sick Leave / Q000191:economics; D018434:Sickness Impact Profile; D012959:Socioeconomic Factors; D011795:Surveys and Questionnaires; D006113:United Kingdom; D055815:Young Adult
keywords: Costs; Costs of illness; Juvenile idiopathic arthritis; Quality of life; Rare diseases