Disease-Associated Costs in Children With Inflammatory Bowel Disease: A Systematic Review

Abstract

Abstract Background As a chronic noncurable disorder often diagnosed in childhood or adolescence, inflammatory bowel disease (IBD) confers a significant financial lifetime burden. The objective of this systematic review was to determine the disease-associated costs (both direct and indirect) associated with IBD in children and young adults. Methods We conducted a systematic review of the literature and included any study reporting direct health services–related costs or the indirect economic burden of IBD in persons aged ≤19 years (PROSPERO protocol number CRD2016036128). A technical panel of experts in pediatric gastroenterology and research methodology formulated the review questions, reviewed the search strategies and review methods, and provided input throughout the review process. Results Nine studies met criteria for inclusion, 6 of which examined direct costs, 1 of which examined both direct and indirect costs, 1 of which assessed indirect costs, and 1 of which assessed out-of-pocket (OOP) costs. Inflammatory bowel disease–associated costs were significantly higher compared with costs in non-IBD populations, with wide variations in cost estimates, which prevented us from conducting a meta-analysis. Costs in Crohn’s disease were higher than in ulcerative colitis. Overall, direct costs shifted from inpatient hospitalization as a major source of direct costs to medications, mainly driven by anti–tumor necrosis factor agents, as the leading cause of direct costs. Predictors of high costs included uncontrolled disease, corticosteroid treatment in the previous year, and comorbidity burden. Conclusions The pediatric literature examining IBD-attributable costs is limited, with widely variable cost estimates. There is a significant knowledge gap in the research surrounding indirect costs and OOP expenses.