OBJECTIVES

Juvenile idiopathic arthritis (JIA) is the most common pediatric rheumatologic disease and can result in life-long disability. New treatments have the potential to alter disease course, however at a potential high cost. Cost-utility analysis (CUA) weighs costs and benefits of interventions to inform decision-making, and is the type of economic evaluation recommended by most HTA guidelines. Health state utility values (HSUV) are used to calculate quality-adjusted life-years (QALYs), the outcome used in CUAs to assess benefit. This systematic review aims to identity papers reporting HSUV to inform parameter inputs for model-based CUA for JIA treatment.

METHODS

Medline, EMBASE, PsyINFO, EconLit and CINAHL were searched (to Nov 2018). Inclusion criteria: English language, reporting primary data using direct or indirect measures of HSUV in children or adults. Abstracts and full-texts were screened independently by two reviewers. A descriptive analysis was conducted.

RESULTS

From 201 identified articles, eleven publications comprising seven unique studies were included. Four studies targeted children, and three reported on adults with JIA. Children’s HSUV were assessed using standard gamble, HUI-3, EQ5D-Y, and EQ5D-3L. Two studies elicited self-reported HSUV from children as well as parent-proxy, and two reported only parent-proxy values. Only one study reported results stratified by disease severity. However, HSUV were not distinct between groups. Overall, in the four studies, HSUV ranged from 0.53 to 0.95. Adult HSUV were measured using EQ5D-3L in all studies and ranged from 0.26 to 0.90. Only one study stratified the results by the presence of uveitis; however, HSUV were not different between groups.

CONCLUSIONS

Few studies reporting HSUV were identified, and the majority did not stratify HSUV to allow parameter input for different health states. Available data are not sufficient to inform short-term or life-time horizon models. There remains urgent need for rigorous HSUV assessment to inform model-based CUAs in JIA.