OBJECTIVE: In the United States, congenital cytomegalovirus disease (cCMVd) is a major cause of permanent disabilities, and remains the most common etiology of non-genetic sensorineural hearing loss. Strategies aimed at prevention are in development, and their evaluation will require estimates of the economic implications of cCMVd. The objective of this project was to develop a conceptual framework to characterize the lifetime economic burden of cCMVd in the US, and to use that framework to identify knowledge gaps.
METHODS: After a targeted literature review, an initial framework was constructed, which was subsequently validated and refined in consultation with CMV experts and stakeholders. The framework was constructed as a brace diagram, which groups clinical events to create distinct patient profiles, each associated with a unique economic trajectory. Published direct healthcare, direct non-healthcare, indirect, and intangible costs were then identified to populate the framework.
RESULTS: cCMVd profiles were stratified by diagnosis/intervention at birth and type of permanent disability(ies), with the distribution and magnitude of costs varying by patient life stage (e.g., neonatal, childhood, adolescence, early adulthood, and late adulthood). However, the framework could not be populated based solely on published cost analyses. Direct healthcare costs associated with the neonatal period, and screening and management of disabilities during early childhood, were the components best characterized in the literature. Gaps were identified in costs associated with adulthood and with severe impairment, and across all life stages, for direct non-healthcare, indirect, and intangible costs.
CONCLUSIONS: The lifetime economic burden of cCMVd in the US has not been documented in the literature. This framework provides the basis for a research agenda and data analysis addressing key knowledge gaps. A full understanding of the lifetime economic burden of cCMVd would inform researchers, policymakers, and other stakeholders as they assess the potential value of cCMVd prevention.
