This study aimed to provide a comprehensive overview of cost indicators and outcome measures used to measure financial burden in families of children with life-limiting conditions.
A scoping review methodology was used to map the existing literature and provide an overview of available cost indicators and outcome measures. Key medical, economic, and scientific databases were systematically searched to identify relevant articles published in 2000 or later.
The database search yielded 7194 records, including 30 articles eligible for final inclusion. Retrieved cost indicators and outcome measures fell into 3 broad categories: direct costs, indirect costs, and financial support. No study comprehensively assessed all 3 categories. Cost indicators used to measure direct costs were grouped into 5 medical and 11 nonmedical out-of-pocket expenses categories, of which 5 were commonly assessed (ie, treatment and diagnostics, travel and transport, accommodation, food, childcare and home help). Half of the reviewed studies included assessments of indirect costs, most commonly estimating work-related income loss by evaluating employment disruptions. Assessments of opportunity costs arising from informal caregiving and of financial support were rarely included.
Current estimates of the financial burden faced by families of children with life-limiting conditions are inconsistent and often incomplete, likely resulting in severe underestimations of the costs these families incur. We hope that the framework presented in this article will contribute to a more comprehensive assessment of illness-related financial burden and help guide future policies in this area.