Abstract

BackgroundThis study aims to describe current practice in identifying and measuring health care resource use and unit costs in economic evaluations or costing studies of juvenile idiopathic arthritis (JIA).MethodsA scoping review was conducted (in July 2018) in PubMed and Embase to identify economic evaluations, costing studies, or resource utilization studies focusing on patients with JIA. Only English language peer-reviewed articles reporting primary research were included. Data from all included full-text articles were extracted and analysed to identify the reported health care resource use items. In addition, the data sources used to obtain these resource use and unit costs were identified for all included articles.ResultsOf 1176 unique citations identified by the search, 20 full-text articles were included. These involved 4 full economic evaluations, 5 cost-outcome descriptions, 8 cost descriptions, and 3 articles reporting only resource use. The most commonly reported health care resource use items involved medication (80%), outpatient and inpatient hospital visits (80%), laboratory tests (70%), medical professional visits (70%) and other medical visits (65%). Productivity losses of caregivers were much more often incorporated than (future) productivity losses of patients (i.e. 55% vs. 15%). Family borne costs were not commonly captured (ranging from 15% for school costs to 50% for transportation costs). Resource use was mostly obtained from family self-reported questionnaires. Estimates of unit costs were mostly based on reimbursement claims, administrative data, or literature.ConclusionsDespite some consistency in commonly included health care resource use items, variability remains in including productivity losses, missed school days and family borne costs. As these items likely substantially influence the full cost impact of JIA, the heterogeneity found between the items reported in the included studies limits the comparability of the results. Therefore, standardization of resource use items and unit costs to be collected is required. This standardization will provide guidance to future research and thereby improve the quality and comparability of economic evaluations or costing studies in JIA and potentially other childhood diseases. This would allow better understanding of the burden of JIA, and to estimate how it varies across health care systems.

Highlights

  • This study aims to describe current practice in identifying and measuring health care resource use and unit costs in economic evaluations or costing studies of juvenile idiopathic arthritis (JIA)

  • A scoping review was conducted to identify resource use items and unit costs included in economic evaluations or costing/resource utilization studies in JIA [26, 27]

  • Conclusions there is some consistency in terms of items that are included in studies examining resource use and unit costs of JIA from the health care system perspective, there is heterogeneity in what is included within perspectives going beyond that

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Summary

Introduction

This study aims to describe current practice in identifying and measuring health care resource use and unit costs in economic evaluations or costing studies of juvenile idiopathic arthritis (JIA). Juvenile idiopathic arthritis (JIA) is the most common chronic rheumatologic disease in childhood [1]. Due to its chronic nature, children diagnosed with JIA are at higher risk of developing behaviour and/or psychiatric disorders (e.g. social isolation, depression, anxiety) [6, 7], they miss more school or work days due to symptoms or medical appointments [8, 9], and have lower Health-Related Quality of Life (HRQoL) [6] than their peers. In patients with active disease or with inadequate response to conventional pharmacological treatment (e.g. MTX, NSAIDs and DMARDs), the use of a (much more expensive) biologic DMARD is recommended [4, 10, 11]

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