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Abstract
BackgroundIn the United States (US), congenital cytomegalovirus infection (cCMVi) is a major cause of permanent disabilities and the most common etiology of non-genetic sensorineural hearing loss. Evaluations of prevention strategies will require estimates of the economic implications of cCMVi. We aimed to develop a conceptual framework to characterize the lifetime economic burden of cCMVi in the US and to use that framework to identify data gaps.MethodsDirect health care, direct non-health care, indirect, and intangible costs associated with cCMVi were considered. An initial framework was constructed based on a targeted literature review, then validated and refined after consultation with experts. Published costs were identified and used to populate the framework. Data gaps were identified.ResultsThe framework was constructed as a chance tree, categorizing clinical event occurrence to form patient profiles associated with distinct economic trajectories. The distribution and magnitude of costs varied by patient life stage, cCMVi diagnosis, severity of impairment, and developmental delays/disabilities. Published studies could not fully populate the framework. The literature best characterized direct health care costs associated with the birth period. Gaps existed for direct non-health care, indirect, and intangible costs, as well as health care costs associated with adult patients and those severely impaired.ConclusionsData gaps exist concerning the lifetime economic burden of cCMVi in the US. The conceptual framework provides the basis for a research agenda to address these gaps. Understanding the full lifetime economic burden of cCMVi would inform clinicians, researchers, and policymakers, when assessing the value of cCMVi interventions.
Highlights
In the United States (US), congenital cytomegalovirus infection is a major cause of permanent disabilities and the most common etiology of non-genetic sensorineural hearing loss
The virus is spread by close contact with an infected person through saliva, urine, or other body fluids and can be transmitted from a pregnant woman to her fetus during pregnancy—termed congenital CMV infection
Conceptual framework Key articles contributing insights to the development of the conceptual framework included cost-effectiveness analyses of interventions or vaccines for congenital cytomegalovirus infection (cCMVi) in the US [22,23,24,25]; most of these analyses [22,23,24] relied on data for non-cCMVi-specific patient populations with disabilities that were published more than 25 years ago
Summary
In the United States (US), congenital cytomegalovirus infection (cCMVi) is a major cause of permanent disabilities and the most common etiology of non-genetic sensorineural hearing loss. 50% of people in the United States (US) have had human cytomegalovirus (CMV) infection by adulthood, as determined by positive serology [1]. About 25% of infants with cCMVi are born with abnormalities such as sensorineural hearing loss (SNHL), chorioretinitis, jaundice, hepatitis, or microcephaly [2, 4]. Regardless of clinical status at birth, approximately 18% of all children with cCMVi develop permanent neurodevelopmental delays or disabilities, often associated with SNHL or vision loss [4]. With cCMVi being the most common congenital infection in the US [5, 6], a significant need exists to reduce its burden
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