Economic evaluation of newborn screening for congenital cytomegalovirus infection: A systematic review

Eur J Pediatr. 2025 Jan 9;184(1):123. doi: 10.1007/s00431-024-05953-1.

Abstract

Purpose: This systematic review analyzes economic evaluations of newborn screening for congenital cytomegalovirus (cCMV) infection to identify key factors influencing cost-effectiveness and differences in methodological approaches.

Methods: Following a pre-registered PROSPERO protocol (CRD42023441587), we conducted a comprehensive literature search across multiple databases on July 4, 2024. The review included both full economic evaluations (cost and outcomes) and partial economic evaluations (cost only). Two reviewers independently performed data extraction and synthesis. Quality assessment used the Consensus Health Economic Criteria extended checklist and the Consolidated Health Economic Evaluation Reporting Standards 2022 Statement.

Results: From 543 records, nine studies met inclusion criteria: four full and five partial economic evaluations. Two full economic evaluations provided the incremental cost-effectiveness ratio per quality-adjusted life year. Full economic evaluations using decision trees and Markov models generally found universal screening more cost-effective than targeted screening, despite higher incremental costs. Partial evaluations focused on direct costs, with varying inclusion of long-term care costs. Most studies adopted a healthcare system perspective, excluding indirect costs. Quality assessment of full economic evaluations revealed high methodological standards but identified common limitations in outcome measurement and reporting.

Conclusions: Available evidence suggests potential cost-effectiveness of newborn cCMV screening, particularly with early intervention strategies. However, limited full economic evaluations and heterogeneous methodological approaches preclude definitive conclusions, highlighting the need for additional research across diverse healthcare contexts.

What is known: • Congenital cytomegalovirus (cCMV) infection is a condition that can be detected through newborn screening, and understanding its economic implications is important for healthcare systems. • Economic evaluations can be categorized into full evaluations (analyzing both costs and outcomes) and partial evaluations (focusing only on costs).

What is new: • Based on a systematic review of 543 records, universal screening appears more cost-effective than targeted screening for cCMV, implementation challenges and healthcare system variations significantly impact its practical adoption. • The review identified significant methodological limitations in existing studies, particularly in outcome measurement and reporting, highlighting the need for more comprehensive research across different healthcare contexts.

Keywords: Congenital cytomegalovirus infection; Cost-effectiveness; Economic evaluation; Screening.

Publication types

  • Systematic Review
  • Review

MeSH terms

  • Cost-Benefit Analysis*
  • Cytomegalovirus Infections* / congenital
  • Cytomegalovirus Infections* / diagnosis
  • Cytomegalovirus Infections* / economics
  • Humans
  • Infant, Newborn
  • Neonatal Screening* / economics
  • Neonatal Screening* / methods
  • Quality-Adjusted Life Years