Seeking the state of the art in standardized measurement of health care resource use and costs in juvenile idiopathic arthritis: A scoping review

Michelle M.A. Kip, Gillian Currie, Deborah A. Marshall, Luiza Grazziotin Lago, Marinka Twilt, Sebastiaan J. Vastert, Joost F. Swart, Nico Wulffraat, Rae S.M. Yeung, Susanne M. Benseler, Maarten J. Ijzerman*, Gouke Bonsel, Brian M. Feldman, Esther Hoppenreijs, Bianca Lang, Claire Leblanc, Ana Sepulveda, Karine Toupin-April, Philomine Van Pelt, Annet Van Royen-Kerkhof

*Corresponding author for this work

Research output: Contribution to journalReview articlepeer-review


Background: 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). Methods: A 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. Results: Of 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. Conclusions: Despite 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.

Original languageEnglish
Article number20
Number of pages13
JournalPediatric Rheumatology
Issue number1
Publication statusPublished - 6 May 2019


  • Ambulatory Care/economics
  • Arthritis, Juvenile/economics
  • Caregivers/economics
  • Child
  • Clinical Laboratory Techniques/economics
  • Efficiency
  • Facilities and Services Utilization
  • Health Care Costs/statistics & numerical data
  • Health Resources/economics
  • Hospitalization/economics
  • Humans


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