Skip to main content
Log in

Health State Utility Values in Juvenile Idiopathic Arthritis: What is the Evidence?

  • Systematic Review
  • Published:
PharmacoEconomics Aims and scope Submit manuscript

Abstract

Objectives

The objectives of this systematic review were to identify health state utility values (HSUV) of children and adults with juvenile idiopathic arthritis in the literature and to assess whether HSUV were appropriately reported and could be used to inform parameter inputs for a model-based cost-utility analysis to inform decision making.

Methods

MEDLINE, EMBASE, PsycINFO, EconLit and CINAHL databases were searched in July 2019. Inclusion criteria were studies using preference-based instruments, targeting children or adults with juvenile idiopathic arthritis, and in the English language. The quality of studies was assessed using a modified checklist that included relevant sources of bias and assessment of quality of HSUV valuation and measurement. A descriptive analysis was conducted, including assessment on reporting of population characteristics and stratification of HSUV by potential health states or population subgroup.

Results

From 620 identified articles, ten reported HSUV. Seven studies reported HSUV of children with juvenile idiopathic arthritis, and three of adults with a history of juvenile idiopathic arthritis. Population disease activity status and drug treatment were reported in less than half of the studies. Six (out of ten) studies stratified HSUV results for at least one of the potential health state categories, but they represent very specific situations or interventions (e.g. patients receiving different types of physiotherapy or treated with etanercept over time).

Conclusions

We have identified critical gaps in the literature reporting HSUV in patients with juvenile idiopathic arthritis including a lack of HSUV measures for distinct health states, particularly in adults with a history of juvenile idiopathic arthritis. The current reported HSUV data in juvenile idiopathic arthritis are insufficient for a full cost-utility analysis with a short or lifetime horizon.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2
Fig. 3

Similar content being viewed by others

Data Availability

The detailed information regarding search strategy, data extracted from included studies and quality assessment of individual studies are available in the Electronic Supplementary Material.

References

  1. Prakken B, Albani S, Martini A. Juvenile idiopathic arthritis. Lancet. 2011;377(9783):2138–49. https://doi.org/10.1016/s0140-6736(11)60244-4.

    Article  PubMed  Google Scholar 

  2. Shiff NJ, Oen K, Kroeker K, Lix LM. Trends in population-based incidence and prevalence of juvenile idiopathic arthritis in Manitoba, Canada. Arthritis Care Res. 2019;71(3):413–8. https://doi.org/10.1002/acr.23606.

    Article  Google Scholar 

  3. Kip MMA, Currie G, Marshall DA, Grazziotin Lago L, Twilt M, Vastert SJ, et al. Seeking the state of the art in standardized measurement of health care resource use and costs in juvenile idiopathic arthritis: a scoping review. Pediatr Rheumatol Online J. 2019;17(1):20. https://doi.org/10.1186/s12969-019-0321-x.

    Article  PubMed  PubMed Central  Google Scholar 

  4. Tollisen A, Selvaag AM, Aulie HA, Lilleby V, Aasland A, Lerdal A, et al. Physical functioning, pain, and health-related quality of life in adults with juvenile idiopathic arthritis: a longitudinal 30-year followup study. Arthritis Care Res. 2018;70(5):741–9. https://doi.org/10.1002/acr.23327.

    Article  Google Scholar 

  5. Oliveira-Ramos F, Eusebio M, Mourao AF, Furtado C, Campanilho-Marques R, et al. Juvenile idiopathic arthritis in adulthood: fulfilment of classification criteria for adult rheumatic diseases, long-term outcomes and predictors of inactive disease, functional status and damage. RMD Open. 2016;2(2):e000304. https://doi.org/10.1136/rmdopen-2016-000304.

    Article  PubMed  PubMed Central  Google Scholar 

  6. Albers HM, Wessels JA, van der Straaten RJ, Brinkman DM, Suijlekom-Smit LW, Kamphuis SS, et al. Time to treatment as an important factor for the response to methotrexate in juvenile idiopathic arthritis. Arthritis Rheum. 2009;61(1):46–51. https://doi.org/10.1002/art.24087.

    Article  CAS  PubMed  Google Scholar 

  7. Otten MH, Anink J, Prince FH, Twilt M, Vastert SJ, ten Cate R, et al. Trends in prescription of biological agents and outcomes of juvenile idiopathic arthritis: results of the Dutch National Arthritis and Biologics in Children Register. Ann Rheum Dis. 2015;74(7):1379–86. https://doi.org/10.1136/annrheumdis-2013-204641.

    Article  PubMed  Google Scholar 

  8. Brazier J. Valuing health states for use in cost-effectiveness analysis. Pharmacoeconomics. 2008;26(9):769–79. https://doi.org/10.2165/00019053-200826090-00007.

    Article  PubMed  Google Scholar 

  9. Brazier J, Ara R, Azzabi I, Busschbach J, Chevrou-Severac H, Crawford B, et al. Identification, review, and use of health state utilities in cost-effectiveness models: an ISPOR Good Practices for Outcomes Research Task Force Report. Value Health. 2019;22(3):267–75. https://doi.org/10.1016/j.jval.2019.01.004.

    Article  PubMed  Google Scholar 

  10. Roberts M, Russell LB, Paltiel AD, Chambers M, McEwan P, Krahn M. Conceptualizing a model: a report of the ISPOR-SMDM Modeling Good Research Practices Task Force-2. Med Decis Mak. 2012;32(5):678–89. https://doi.org/10.1177/0272989x12454941.

    Article  Google Scholar 

  11. Caro JJ, Briggs AH, Siebert U, Kuntz KM. Modeling good research practices: overview. A report of the ISPOR-SMDM Modeling Good Research Practices Task Force: 1. Value Health. 2012;15(6):796–803. https://doi.org/10.1016/j.jval.2012.06.012.

    Article  PubMed  Google Scholar 

  12. Petrou S, Kwon J, Madan J. A practical guide to conducting a systematic review and meta-analysis of health state utility values. Pharmacoeconomics. 2018;36(9):1043–61. https://doi.org/10.1007/s40273-018-0670-1.

    Article  PubMed  Google Scholar 

  13. Luca NJ, Burnett HF, Ungar WJ, Moretti ME, Beukelman T, Feldman BM, et al. Cost-effectiveness analysis of first-line treatment with biologic agents in polyarticular juvenile idiopathic arthritis. Arthritis Care Res. 2016;68(12):1803–11.

    Article  CAS  Google Scholar 

  14. Ungar WJ, Costa V, Hancock-Howard R, Feldman BM, Laxer RM. Cost-effectiveness of biologics in polyarticular-course juvenile idiopathic arthritis patients unresponsive to disease-modifying antirheumatic drugs. Arthritis Care Res. 2011;63(1):111–9.

    Article  Google Scholar 

  15. Kwon J, Kim SW, Ungar WJ, Tsiplova K, Madan J, Petrou S. A systematic review and meta-analysis of childhood health utilities. Med Decis Mak. 2018;38(3):277–305. https://doi.org/10.1177/0272989x17732990.

    Article  Google Scholar 

  16. Shepherd J, Cooper K, Harris P, Picot J, Rose M. The clinical effectiveness and cost-effectiveness of abatacept, adalimumab, etanercept and tocilizumab for treating juvenile idiopathic arthritis: a systematic review and economic evaluation. Health Technol Assess. 2016;20(34):1–222.

    Article  PubMed  PubMed Central  Google Scholar 

  17. Bansback N, Ara R, Karnon J, Anis A. Economic evaluations in rheumatoid arthritis: a critical review of measures used to define health States. Pharmacoeconomics. 2008;26(5):395–408. https://doi.org/10.2165/00019053-200826050-00004.

    Article  PubMed  Google Scholar 

  18. Ara R, Brazier J, Peasgood T, Paisley S. The identification, review and synthesis of health state utility values from the literature. Pharmacoeconomics. 2017;35(Suppl. 1):43–55. https://doi.org/10.1007/s40273-017-0547-8.

    Article  PubMed  Google Scholar 

  19. Brunner HI, Klein-Gitelman MS, Miller MJ, Trombley M, Baldwin N, Kress A, et al. Health of children with chronic arthritis: relationship of different measures and the quality of parent proxy reporting. Arthritis Rheum. 2004;51(5):763–73.

    Article  PubMed  Google Scholar 

  20. Prince FH, de Bekker-Grob EW, Twilt M, van Rossum MA, Hoppenreijs EP, ten Cate R, et al. An analysis of the costs and treatment success of etanercept in juvenile idiopathic arthritis: results from the Dutch Arthritis and Biologicals in Children Register. Rheumatology. 2011;50(6):1131–6.

    Article  CAS  PubMed  Google Scholar 

  21. Anink J, Prince FH, Dijkstra M, Otten MH, Twilt M, ten Cate R, et al. Long-term quality of life and functional outcome of patients with juvenile idiopathic arthritis in the biologic era: a longitudinal follow-up study in the Dutch Arthritis and Biologicals in Children Register. Rheumatology. 2015;54(11):1964–9.

    Article  PubMed  Google Scholar 

  22. Prince FH, Geerdink LM, Borsboom GJ, Twilt M, van Rossum MA, Hoppenreijs EP, et al. Major improvements in health-related quality of life during the use of etanercept in patients with previously refractory juvenile idiopathic arthritis. Ann Rheum Dis. 2010;69(1):138–42.

    Article  CAS  PubMed  Google Scholar 

  23. 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 idiopathic arthritis. Health Technol Assess. 2005;9(39):iii–iv, ix–x, 1–59.

  24. Hendry GJ, Watt GF, Brandon M, Friel L, Turner DE, Lorgelly PK, et al. The effectiveness of a multidisciplinary foot care program for children and adolescents with juvenile idiopathic arthritis: an exploratory trial. J Rehabil Med. 2013;45(5):467–76. https://doi.org/10.2340/16501977-1130.

    Article  PubMed  Google Scholar 

  25. Ramanan AV, Dick AD, Jones AP, Hughes DA, McKay A, Rosala-Hallas A, et al. Adalimumab in combination with methotrexate for refractory uveitis associated with juvenile idiopathic arthritis: a RCT. Health Technol Assess. 2019;23(15):1–140. https://doi.org/10.3310/hta23150.

    Article  PubMed  PubMed Central  Google Scholar 

  26. Barth S, Haas JP, Schlichtiger J, Molz J, Bisdorff B, Michels H, et al. Long-term health-related quality of life in German patients with juvenile idiopathic arthritis in comparison to German general population. PLoS ONE. 2016;11(4):e0153267.

    Article  PubMed  PubMed Central  Google Scholar 

  27. Haasnoot AJW, Sint Jago NFM, Tekstra J, de Boer JH. Impact of uveitis on quality of life in adult patients with juvenile idiopathic arthritis. Arthritis Care Res. 2017;69(12):1895–902.

    Article  CAS  Google Scholar 

  28. Mittmann N, Trakas K, Risebrough N, Liu BA. Utility scores for chronic conditions in a community-dwelling population. Pharmacoeconomics. 1999;15(4):369–76. https://doi.org/10.2165/00019053-199915040-00004.

    Article  CAS  PubMed  Google Scholar 

  29. Petrou S, Kupek E. Estimating preference-based Health Utilities Index Mark 3 utility scores for childhood conditions in England and Scotland. Med Decis Mak. 2009;29(3):291–303. https://doi.org/10.1177/0272989x08327398.

    Article  Google Scholar 

  30. Kuhlmann A, Schmidt T, Treskova M, López-Bastida J, Linertová R, Oliva-Moreno J, et al. Social/economic costs and health-related quality of life in patients with juvenile idiopathic arthritis in Europe. Eur J Health Econ. 2016;17:79–877. https://doi.org/10.1007/s10198-016-0786-1.

    Article  PubMed  Google Scholar 

  31. Angelis A, Kanavos P, Lopez-Bastida J, Linertova R, Serrano-Aguilar P. Socioeconomic costs and health-related quality of life in juvenile idiopathic arthritis: a cost-of-illness study in the United Kingdom. BMC Musculoskelet Disord. 2016;17:321.

    Article  PubMed  PubMed Central  Google Scholar 

  32. Payakachat N, Ali MM, Tilford JM. Can the EQ-5D detect meaningful change? A systematic review. Pharmacoeconomics. 2015;33(11):1137–54. https://doi.org/10.1007/s40273-015-0295-6.

    Article  PubMed  PubMed Central  Google Scholar 

  33. Ara R, Rowen D, Mukuria C. The use of mapping to estimate health state utility values. Pharmacoeconomics. 2017;35(Suppl. 1):57–66. https://doi.org/10.1007/s40273-017-0548-7.

    Article  PubMed  Google Scholar 

  34. Feeny D, Furlong W, Torrance GW, Goldsmith CH, Zhu Z, DePauw S, et al. Multiattribute and single-attribute utility functions for the Health Utilities Index Mark 3 system. Med Care. 2002;40(2):113–28.

    Article  PubMed  Google Scholar 

  35. Wille N, Badia X, Bonsel G, Burstrom K, Cavrini G, Devlin N, et al. Development of the EQ-5D-Y: a child-friendly version of the EQ-5D. Qual Life Res. 2010;19(6):875–86. https://doi.org/10.1007/s11136-010-9648-y.

    Article  PubMed  PubMed Central  Google Scholar 

  36. Rowen D, Rivero-Arias O, Devlin N, Ratcliffe J. Review of valuation methods of preference-based measures of health for economic evaluation in child and adolescent populations: where are we now and where are we going? Pharmacoeconomics. 2020;38(4):325–40. https://doi.org/10.1007/s40273-019-00873-7.

    Article  PubMed  Google Scholar 

  37. Kind P, Klose K, Gusi N, Olivares PR, Greiner W. Can adult weights be used to value child health states? Testing the influence of perspective in valuing EQ-5D-Y. Qual Life Res. 2015;24(10):2519–39. https://doi.org/10.1007/s11136-015-0971-1.

    Article  PubMed  PubMed Central  Google Scholar 

  38. Herdman M, Gudex C, Lloyd A, Janssen M, Kind P, Parkin D, et al. Development and preliminary testing of the new five-level version of EQ-5D (EQ-5D-5L). Qual Life Res. 2011;20(10):1727–36. https://doi.org/10.1007/s11136-011-9903-x.

    Article  CAS  PubMed  PubMed Central  Google Scholar 

  39. Buchholz I, Janssen MF, Kohlmann T, Feng Y-S. A systematic review of studies comparing the measurement properties of the three-level and five-level versions of the EQ-5D. Pharmacoeconomics. 2018;36(6):645–61. https://doi.org/10.1007/s40273-018-0642-5.

    Article  PubMed  PubMed Central  Google Scholar 

  40. Conner-Spady BL, Marshall DA, Bohm E, Dunbar MJ, Loucks L, Al Khudairy A, et al. Reliability and validity of the EQ-5D-5L compared to the EQ-5D-3L in patients with osteoarthritis referred for hip and knee replacement. Qual Life Res. 2015;24(7):1775–844. https://doi.org/10.1007/s11136-014-0910-6.

    Article  PubMed  Google Scholar 

  41. Jin X, Al Sayah F, Ohinmaa A, Marshall DA, Smith C, Johnson JA. The EQ-5D-5L is superior to the -3L version in measuring health-related quality of life in patients awaiting THA or TKA. Clin Orthop Relat Res. 2019;477(7):1632–44. https://doi.org/10.1097/corr.0000000000000662.

    Article  PubMed  PubMed Central  Google Scholar 

  42. Weinstein MC, Torrance G, McGuire A. QALYs: the basics. Value Health. 2009;12(Suppl. 1):S5–9. https://doi.org/10.1111/j.1524-4733.2009.00515.x.

    Article  PubMed  Google Scholar 

  43. Ara R, Brazier J, Zouraq IA. The use of health state utility values in decision models. Pharmacoeconomics. 2017;35(Suppl. 1):77–88. https://doi.org/10.1007/s40273-017-0550-0.

    Article  PubMed  Google Scholar 

Download references

Acknowledgements

We acknowledge the contributions of Carolina de La Rosa as the second reviewer for title and abstract screening. This study is part of the Understanding Childhood Arthritis Network Canada-Netherlands (UCAN CAN-DU) and UCAN CURE program of work, and we acknowledge the UCAN CAN-DU and UCAN CURE leadership team (alphabetical order): Susanne M. Benseler, Maarten Ijzerman, Deborah A Marshall, Joost F. Swart, Marinka Twilt, Sebastiaan J. Vastert, Nico Wulffraat and Rae S.M. Yeung.

Author information

Authors and Affiliations

Authors

Contributions

All authors contributed to the systematic review conception and design, and to the drafting of this manuscript. LRG and RL performed full-text screening, data extraction and quality assessment of included papers. Expert guidance was provided by DM throughout all steps of the manuscript development process. DAM, MJI, MT, MMAK and GC provided expert guidance on assessing the appropriateness of health state utility values and interpreting the findings. All named authors meet the International Committee of Medical Journal Editors criteria for authorship for this manuscript, take responsibility for the integrity of the work as a whole and have given final approval to the version to be published.

Corresponding author

Correspondence to Deborah A. Marshall.

Ethics declarations

Funding

This work was supported by the Canadian Institutes for Health Research (Canada) [grant number 381280]; Genome Canada (Canada); ZonMW (the Netherlands); and the Reumafonds (the Netherlands). Deborah A. Marshall is supported by the Arthur J.E. Child Chair in Rheumatology and a Canada Research Chair in Health Systems and Services Research (2008–2018). Luiza R. Grazziotin was funded by the Arthritis Society (TGP-18-0244) and UCAN CAN-DU project.

Conflict of interest

Luiza R. Grazziotin, Gillian Currie, Michelle M. A. Kip, Maarten J. IJzerman, Marinka Twilt, Raymond Lee, and Deborah A. Marshall have no conflicts of interest that are directly relevant to the content of this article.

Additional information

The core investigators of UCAN CAN-DU team are listed in the Acknowledgements section.

Electronic supplementary material

Below is the link to the electronic supplementary material.

Supplementary file1 (DOCX 43 kb)

Rights and permissions

Reprints and permissions

About this article

Check for updates. Verify currency and authenticity via CrossMark

Cite this article

Grazziotin, L.R., Currie, G., Kip, M.M.A. et al. Health State Utility Values in Juvenile Idiopathic Arthritis: What is the Evidence?. PharmacoEconomics 38, 913–926 (2020). https://doi.org/10.1007/s40273-020-00921-7

Download citation

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s40273-020-00921-7

Navigation