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. Inclusion criteria: studies reporting HSUV, targeting children or adults with JIA, and in English language. Abstracts and full-texts were screened independently by two reviewers. A descriptive analysis was conducted, including assessment on whether studies report HSUV stratified by potential treatment-related health states (e.g. disease activity, treatment status) or population subgroup.

RESULTS

From 324 identified articles, nine publications were included. Six studies targeted children, and three reported on adults with history of JIA. Children’s HSUV were assessed using standard gamble, HUI-3, EQ5D-Y, and EQ5D-3L. Three studies elicited self-reported HSUV from children, and three reported only parent-proxy values. One study reported HSUV for etanercept treatment, and one reported HSUV stratified by disease severity. However, in the latter, HSUV were not distinct between groups. Adult HSUV were measured using EQ5D-3L in all studies. Only one study stratified HSUV by the presence of uveitis, but found no difference between groups.

CONCLUSIONS

Few studies reporting HSUV were identified, and the majority did not stratify HSUV to allow parameter input for different treatment-related 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.