A systematic literature review of economic evaluations and cost-of-illness studies of inherited cardiomyopathies

Hypertrophic cardiomyopathy (HCM) and dilated cardiomyopathy (DCM) are commonly inherited heart conditions associated with a high risk of heart failure and sudden cardiac death. To understand the economic and societal disease burden, this study systematically identified and reviewed cost-of-illness (COI) studies and economic evaluations (EEs) of various interventions for HCM and DCM. A literature search was performed in MEDLINE, EMBASE, NHS EED, EconLit and Web of Science to identify COI studies and EEs published between 1 January 2010 and 28 April 2021. The selection of studies and their critical appraisal were performed jointly by two independent researchers. For the quality assessment, the ‘Consensus on Health Economic Criteria’ list was used. Two COI studies and 11 EEs were eligible for inclusion. Cost-effectiveness varied among interventions and depended on the targeted patient population. Both COI studies identified only hospitalisation costs in HCM. The mean study quality was high in EEs but low in COI studies. Most studies excluded costs for patients, caregivers and productivity losses. Overall, knowledge of the societal and economic burden of inherited cardiomyopathies is limited. Future research needs to include quality-adjusted life years and a broader range of costs to provide an information base for optimising care for affected patients.