The cost-effectiveness of targeted screening for congenital cytomegalovirus in newborns compared to clinical diagnosis in the US

Congenital cytomegalovirus (cCMV) is the leading environmental cause of hearing loss (HL) among children, affecting four in one thousand newborns. cCMV testing in the US is currently based on clinical diagnosis which does not consistently identify cCMV cases and precludes early intervention to prevent and reduce the severity of HL. We estimated the cost-effectiveness of targeted newborn screening and cCMV testing among newborns compared to clinical diagnosis. We use a decision-analytic model to estimate the costs of preventing HL progression, of additional cases of severe HL, of identifying a case of HL one year earlier, and of identifying an additional case of cCMV, through targeted screening and cCMV testing for infants failing two newborn hearing screens with follow-up to age five. We also estimate the costs of nationwide implementation of a newborn screening and testing program. Model pathways were based on best practices for screening, testing, and treatment. Probabilities were drawn from the published literature; costs were estimated based on Medicare reimbursement rates. Probabilistic and scenario analyses were conducted to determine the robustness of results. Targeted testing and cCMV screening, compared to standard of care, cost an additional $2.96 (±2.26) per infant screened and identified 0.00038 (±0.00022) cases of HL, 3.8 in 10000 children, at a cost of $8197 (±4217) per case of HL identified. Implementing targeted screening for all children in the US was estimated to cost $193,229. Although cases numbers are small, our model shows that targeted newborn screening and cCMV testing reduced cases of HL progression. Adoption of newborn targeted screening as standard of care should be considered given it may prevent disability at very low cost.