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Published in: BMC Musculoskeletal Disorders 1/2016

Open Access 01-12-2016 | Research article

Socioeconomic costs and health-related quality of life in juvenile idiopathic arthritis: a cost-of-illness study in the United Kingdom

Authors: Aris Angelis, Panos Kanavos, Julio López-Bastida, Renata Linertová, Pedro Serrano-Aguilar, BURQOL-RD Research Network

Published in: BMC Musculoskeletal Disorders | Issue 1/2016

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Abstract

Background

Juvenile idiopathic arthritis (JIA) refers to a number of rare chronic inflammatory diseases. Although JIA imposes a significant societal burden, limited data are available on the cost of JIA. The study’s objective is to quantify the socioeconomic burden of JIA patients in the United Kingdom (UK), along with their health-related quality of life (HRQoL).

Methods

A bottom-up, cross-sectional, cost-of-illness analysis of 23 patients was carried out. To collect data on demographic characteristics, health resource utilization, informal care, productivity losses and HRQoL, questionnaires were administered to and completed by patients or their caregivers. The EuroQol five dimensions (EQ-5D) instrument was used to measure HRQoL.

Results

This study found that the average annual cost for a JIA patient was €31,546, with direct health care costs equalling €14,509 (46.0 % of total costs), direct non-health care costs amounting to €8,323 (26.4 %) and productivity losses being €8,715 (27.6 %). This was calculated using unit costs for 2012. The largest expenditures on average were accounted for by early retirement (27.0 %), followed by informal care (24.1 %), medications (21.1 %), outpatient and primary health care visits (13.2 %) and diagnostic tests (7.9 %). Important differences existed between JIA patients in need of caregiver assistance and those with no need (€39,469 vs. €25,452 respectively). Among adult JIA patients, mean EQ-5D index scores and visual analogue scale (VAS) scores were found to be 0.26 and 49.00 respectively; the same scores among caregivers were 0.66 and 67.14 respectively.

Conclusion

JIA poses a significant cost burden on the UK society. Over half of the total average costs (54 %) are related to non-health care and productivity losses. HRQoL of JIA patients is considerably worse than the UK general population.
Appendix
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Footnotes
1
Such care is provided by non-professional caregivers who are not remunerated, primarily being relatives, friends or neighbours of patients.
 
Literature
1.
go back to reference Petty RE, Southwood TR, Baum J, Bhettay E, Glass DN, Manners P, et al. Revision of the proposed classification criteria for juvenile idiopathic arthritis: Durban, 1997. J Rheumatol. 1998;25:1991–4.PubMed Petty RE, Southwood TR, Baum J, Bhettay E, Glass DN, Manners P, et al. Revision of the proposed classification criteria for juvenile idiopathic arthritis: Durban, 1997. J Rheumatol. 1998;25:1991–4.PubMed
2.
go back to reference Minden K, Niewerth M, Listing J, Biedermann T, Schöntube M, Zink A. Burden and cost of illness in patients with juvenile idiopathic arthritis. Ann Rheum Dis. 2004;63:836–42.CrossRefPubMedPubMedCentral Minden K, Niewerth M, Listing J, Biedermann T, Schöntube M, Zink A. Burden and cost of illness in patients with juvenile idiopathic arthritis. Ann Rheum Dis. 2004;63:836–42.CrossRefPubMedPubMedCentral
3.
go back to reference Helmick CG, Felson DT, Lawrence RC, Gabriel S, Hirsch R, Kwoh CK, et al. Estimates of the prevalence of arthritis and other rheumatic conditions in the United States. Part I Arthritis Rheum. 2008;58:15–25.CrossRefPubMed Helmick CG, Felson DT, Lawrence RC, Gabriel S, Hirsch R, Kwoh CK, et al. Estimates of the prevalence of arthritis and other rheumatic conditions in the United States. Part I Arthritis Rheum. 2008;58:15–25.CrossRefPubMed
4.
go back to reference Symmons DP, Jones M, Osborne J, Sills J, Southwood TR, Woo P. Pediatric rheumatology in the United Kingdom: data from the British pediatric rheumatology group national diagnostic register. J Rheumatol. 1996;23:1975–80.PubMed Symmons DP, Jones M, Osborne J, Sills J, Southwood TR, Woo P. Pediatric rheumatology in the United Kingdom: data from the British pediatric rheumatology group national diagnostic register. J Rheumatol. 1996;23:1975–80.PubMed
5.
go back to reference Huang JL. New advances in juvenile idiopathic arthritis. Chang Gung Med J. 2012;35:1–14.PubMed Huang JL. New advances in juvenile idiopathic arthritis. Chang Gung Med J. 2012;35:1–14.PubMed
6.
go back to reference Weis JE, Ilowite NT. Juvenile idiopathic arthritis. Rheum Dis Clin N Am. 2007;33:441–70.CrossRef Weis JE, Ilowite NT. Juvenile idiopathic arthritis. Rheum Dis Clin N Am. 2007;33:441–70.CrossRef
8.
go back to reference Beukelman T, Patkar NM, Saag KG, Tolleson-Rinehart S, Cron RQ, DeWitt EM, et al. 2011 American College of Rheumatology recommendations for the treatment of juvenile idiopathic arthritis: initiation and safety monitoring of therapeutic agents for the treatment of arthritis and systemic features. Arthritis Care Res. 2011;63:465–82.CrossRef Beukelman T, Patkar NM, Saag KG, Tolleson-Rinehart S, Cron RQ, DeWitt EM, et al. 2011 American College of Rheumatology recommendations for the treatment of juvenile idiopathic arthritis: initiation and safety monitoring of therapeutic agents for the treatment of arthritis and systemic features. Arthritis Care Res. 2011;63:465–82.CrossRef
9.
10.
go back to reference Minden K, Niewerth M, Listing J, Biedermann T, Bollow M, Schöntube M, et al. Long-term outcome in patients with juvenile idiopathic arthritis. Arthritis Rheum. 2002;46:2392–401.CrossRefPubMed Minden K, Niewerth M, Listing J, Biedermann T, Bollow M, Schöntube M, et al. Long-term outcome in patients with juvenile idiopathic arthritis. Arthritis Rheum. 2002;46:2392–401.CrossRefPubMed
14.
go back to reference Bernatsky S, Duffy C, Malleson P, Feldman DE, St Pierre Y, Clarke AE. Economic impact of juvenile idiopathic arthritis. Arthritis Rheum. 2007;57:44–8.CrossRefPubMed Bernatsky S, Duffy C, Malleson P, Feldman DE, St Pierre Y, Clarke AE. Economic impact of juvenile idiopathic arthritis. Arthritis Rheum. 2007;57:44–8.CrossRefPubMed
15.
go back to reference Haverman L, van Oers HA, Maurice-Stam H, Kuijpers TW, Grootenhuis MA, van Rossum MA. Health related quality of life and parental perceptions of child vulnerability among parents of a child with juvenile idiopathic arthritis: results from a web-based survey. Pediatr Rheumatol Online J. 2014;12:34. doi:10.1186/1546-0096-12-34. eCollection 2014.CrossRefPubMedPubMedCentral Haverman L, van Oers HA, Maurice-Stam H, Kuijpers TW, Grootenhuis MA, van Rossum MA. Health related quality of life and parental perceptions of child vulnerability among parents of a child with juvenile idiopathic arthritis: results from a web-based survey. Pediatr Rheumatol Online J. 2014;12:34. doi:10.​1186/​1546-0096-12-34. eCollection 2014.CrossRefPubMedPubMedCentral
16.
go back to reference Linertová R, Serrano-Aguilar P, Posada-de-la-Paz M, Hens-Pérez M, Kanavos P, Taruscio D, on behalf of BURQOL-RD Research Group, et al. Delphi approach to select rare diseases for a European representative survey. The BURQOL-RD study. Health Policy. 2012;108:19–26.CrossRefPubMed Linertová R, Serrano-Aguilar P, Posada-de-la-Paz M, Hens-Pérez M, Kanavos P, Taruscio D, on behalf of BURQOL-RD Research Group, et al. Delphi approach to select rare diseases for a European representative survey. The BURQOL-RD study. Health Policy. 2012;108:19–26.CrossRefPubMed
22.
go back to reference McDaid D. Estimating the costs of informal care for people with Alzheimer’s disease: methodological and practical challenges. Int J of Geriatr Psychiatry. 2001;16:400–5.CrossRef McDaid D. Estimating the costs of informal care for people with Alzheimer’s disease: methodological and practical challenges. Int J of Geriatr Psychiatry. 2001;16:400–5.CrossRef
23.
go back to reference Hodgson TA, Meiners MR. Cost-of-illness methodology: a guide to assessment practices and procedures. Milbank Mem Fund Q. 1982;60:429–91.CrossRef Hodgson TA, Meiners MR. Cost-of-illness methodology: a guide to assessment practices and procedures. Milbank Mem Fund Q. 1982;60:429–91.CrossRef
26.
go back to reference Collin C, Wade DT, et al. The Barthel ADL Index: a reliability study. Int Disabil Stud. 1998;10:61–3.CrossRef Collin C, Wade DT, et al. The Barthel ADL Index: a reliability study. Int Disabil Stud. 1998;10:61–3.CrossRef
27.
go back to reference Hérbert R, Bravo G, Préville M. Reliability, validity, and reference values of the Zarit Burden Interview for assessing informal caregivers of community-dwelling older persons with dementia. Can J Aging. 2000;19:494–507. Hérbert R, Bravo G, Préville M. Reliability, validity, and reference values of the Zarit Burden Interview for assessing informal caregivers of community-dwelling older persons with dementia. Can J Aging. 2000;19:494–507.
29.
go back to reference Kind P, Hardman G, Macran S. UK Population Norms for EQ-5D. Discussion paper 172, 1999. The University of York Centre of Health Economics. Kind P, Hardman G, Macran S. UK Population Norms for EQ-5D. Discussion paper 172, 1999. The University of York Centre of Health Economics.
31.
go back to reference Allaire SH, DeNardo BS, Szer IS, Meenan RF, Schaller JG. The economic impacts of juvenile rheumatoid arthritis. J Rheumatol. 1992;19:952–5.PubMed Allaire SH, DeNardo BS, Szer IS, Meenan RF, Schaller JG. The economic impacts of juvenile rheumatoid arthritis. J Rheumatol. 1992;19:952–5.PubMed
32.
go back to reference Cummins C, Connock M, Fry-Smith A, Burls A. A systematic review of effectiveness and economic evaluation of new drug treatments for juvenile idiopathic arthritis: etanercept. Health Technol Assess. 2002;6:1–43.CrossRefPubMed Cummins C, Connock M, Fry-Smith A, Burls A. A systematic review of effectiveness and economic evaluation of new drug treatments for juvenile idiopathic arthritis: etanercept. Health Technol Assess. 2002;6:1–43.CrossRefPubMed
33.
go back to reference Brunner H, Barron A, Graham T. Effects of treatment on costs &health-related quality of life (HRQL) of children with polyarticularcourse juvenile rheumatoid arthritis (JRA). Arthritis Rheum. 2004;50:S686. Brunner H, Barron A, Graham T. Effects of treatment on costs &health-related quality of life (HRQL) of children with polyarticularcourse juvenile rheumatoid arthritis (JRA). Arthritis Rheum. 2004;50:S686.
34.
go back to reference Minden K, Niewerth M, Listing J, Möbius D, Thon A, Ganser G, et al. The economic burden of juvenile idiopathic arthritis-results from the German paediatric rheumatologic database. Clin Exp Rheumatol. 2009;27:863–9.PubMed Minden K, Niewerth M, Listing J, Möbius D, Thon A, Ganser G, et al. The economic burden of juvenile idiopathic arthritis-results from the German paediatric rheumatologic database. Clin Exp Rheumatol. 2009;27:863–9.PubMed
35.
go back to reference Haapasaari J, Kautiainen HJ, Isomäki HA, Hakala M. Etanercept does not essentially increase the total costs of the treatment of refractory juvenile idiopathic arthritis. J Rheumatol. 2004;31:2286–9.PubMed Haapasaari J, Kautiainen HJ, Isomäki HA, Hakala M. Etanercept does not essentially increase the total costs of the treatment of refractory juvenile idiopathic arthritis. J Rheumatol. 2004;31:2286–9.PubMed
36.
go back to reference Epps H, Ginnelly L, Utley M, Southwood T, Gallivan S, Sculpher M, et al. Is hydrotherapy cost-effective? A randomised controlled trial of combined hydrotherapy programmes compared with physiotherapy land techniques in children with juvenile idiopathicarthritis. Health Technol Assess. 2005;9:1–59.CrossRef Epps H, Ginnelly L, Utley M, Southwood T, Gallivan S, Sculpher M, et al. Is hydrotherapy cost-effective? A randomised controlled trial of combined hydrotherapy programmes compared with physiotherapy land techniques in children with juvenile idiopathicarthritis. Health Technol Assess. 2005;9:1–59.CrossRef
37.
go back to reference April KT, Feldman DE, Platt RW, Duffy CM. Comparison between Children with Juvenile Idiopathic Arthritis (JIA) and their parents concerning perceived Quality of Life. Qual Life Res. 2006;15:655–61.CrossRefPubMed April KT, Feldman DE, Platt RW, Duffy CM. Comparison between Children with Juvenile Idiopathic Arthritis (JIA) and their parents concerning perceived Quality of Life. Qual Life Res. 2006;15:655–61.CrossRefPubMed
38.
go back to reference Shaw KL, Southwood TR, Duffy CM, McDonagh JE. Health-related quality of life in adolescents with juvenile idiopathic arthritis. Arthritis Rheum. 2006;55:199–207.CrossRefPubMed Shaw KL, Southwood TR, Duffy CM, McDonagh JE. Health-related quality of life in adolescents with juvenile idiopathic arthritis. Arthritis Rheum. 2006;55:199–207.CrossRefPubMed
39.
go back to reference Amine B, Rostom S, Benbouazza K, Abouqal R, Hajjaj-Hassouni N. Health related quality of life survey about children and adolescents with juvenile idiopathic arthritis. Rheumatol Int. 2009;29:275–9.CrossRefPubMed Amine B, Rostom S, Benbouazza K, Abouqal R, Hajjaj-Hassouni N. Health related quality of life survey about children and adolescents with juvenile idiopathic arthritis. Rheumatol Int. 2009;29:275–9.CrossRefPubMed
41.
go back to reference Ghatnekar O, Glenngård AH, Olofsson S, Persson U. A literature review of instruments for measuring health-related quality of life in rare diseases. The Swedish Institute for Health Economics. Internal report of BURQOL-RD; 2011. Ghatnekar O, Glenngård AH, Olofsson S, Persson U. A literature review of instruments for measuring health-related quality of life in rare diseases. The Swedish Institute for Health Economics. Internal report of BURQOL-RD; 2011.
Metadata
Title
Socioeconomic costs and health-related quality of life in juvenile idiopathic arthritis: a cost-of-illness study in the United Kingdom
Authors
Aris Angelis
Panos Kanavos
Julio López-Bastida
Renata Linertová
Pedro Serrano-Aguilar
BURQOL-RD Research Network
Publication date
01-12-2016
Publisher
BioMed Central
Published in
BMC Musculoskeletal Disorders / Issue 1/2016
Electronic ISSN: 1471-2474
DOI
https://doi.org/10.1186/s12891-016-1129-1

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