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.
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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.
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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.
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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.
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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.
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The core investigators of UCAN CAN-DU team are listed in the Acknowledgements section.
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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
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DOI: https://doi.org/10.1007/s40273-020-00921-7