Abstract

The aim of this scoping review was to identify current practices in identifying and measuring health care resource use and unit costs in economic evaluation or costing studies of juvenile idiopathic arthritis (JIA). We searched PubMed and EMBASE to August 2017 to identify economic evaluations, costing studies or resource utilization studies focusing on patients with JIA and written in English language. Data from included articles were extracted and analyzed to identify the reported healthcare resource use items and unit costs and the data sources used. Of 1,061 unique citations identified by the search, 20 full-text articles were included. The most commonly reported healthcare resource use items involved medication use (85%), healthcare professional visits (70%) and/or paramedical care (60%), hospitalizations and/or hospital visits (80%), and laboratory tests (75%). Productivity loss of caregivers was considered in the majority studies (65%), but (future) productivity losses of patients were much less commonly considered (35%). Out-of-pocket costs, for example transportation costs, costs of home adaptations, school costs, and the use of complementary and alternative healthcare were only reported in some articles (range 10%-45%). When considering the data sources used, resource use was most frequently obtained from questionnaires. Estimates of unit costs were mostly based on reimbursement claims, administrative data, or the literature. Although there was some consistency in commonly included healthcare resource use items, there remains a gap in reporting items related to productivity losses (lost work and school time) and out-of-pocket costs. However, these are particularly relevant in JIA (and other childhood diseases), because of their strongly debilitating impact and because its consequences may continue into adulthood. Therefore, improved guidance for conducting and reporting (health) economic evaluations for these diseases is required. This can be achieved by developing a standardized list of items for collecting resource use and unit costs in chronic childhood diseases.

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