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

Abstract

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.

Cascade genetic counseling and testing in hereditary syndromes: inherited cardiovascular disease as a model: a narrative review

“…Cascade genetic testing within families with ICDs commenced in the Netherlands in 1996, coinciding with the advent of diagnostic DNA-testing for conditions such as long QT syndrome (LQTS) and hypertrophic cardiomyopathy (HCM) [1]. Since then, cascade genetic testing has emerged as a vital and cost-effective tool for identifying at-risk relatives in cardiogenetics [2][3][4][5][6][7]. It enables relatives with the disease-causing variant to undergo early treatment and prevention efforts, resulting in health benefits and ultimately reducing the cardiovascular burden in the population.…”

Quality of life and societal costs in patients with dilated cardiomyopathy

“… 5 , 6 The disease management of DCM is likely to cause substantial costs for patients and society as a whole; however, the societal and economic burden of DCM remains largely undetermined. 7 …”

“… 10–14 However, available studies are limited and leave out information about costs beyond regular health care, which limits the full picture of the economic disease burden. 7 Given the familial nature of cardiomyopathies, patients might be very young and family members often need to support their relatives, which is likely to cause immense productivity losses and time costs in addition to costs arising for regular medical care.…”

Individual, societal and clinical burden of cardiovascular disease: health technology assessment to guide new diagnostics and treatment strategies

“…which screening strategies, diagnostic strategies and treatment strategies to study, relating to the management of subpopulations of individuals with inherited cardiomyopathy. A systematic literature search indeed illustrated that there is limited current knowledge of the societal and economic burden of the most common forms of inherited cardiac disease: hypertrophic cardiomyopathy (HCM) and dilated cardiomyopathy [ 5 ]. To increase knowledge of QoL and societal costs of HCM in the Netherlands, a prospective burden of disease study in HCM patients and preclinical genotype-positive individuals has been designed [ 6 ].…”