Background

Early detection of cystic fibrosis through newborn screening has significant clinical benefits. Cost-effectiveness plays an important role in selecting the optimal screening strategy from the many available options.

Objectives

The objectives of this study are (1) to summarize study estimates of cost-effectiveness of cystic fibrosis newborn screening (CFNBS) strategies as compared to other strategies, (2) to assess the quality of the studies identified, and (3) to identify determinants of cost-effectiveness.

Methods

Electronic databases were searched from 2007 to June 2017. Health economic evaluations describing the cost-effectiveness of two or more CFNBS strategies were included.

Results

Six health economic evaluations were found. Where included in the comparison, IRT/PAP consistently was the most cost-effective strategy in terms of cost per case detected or life years gained. However, some heterogeneity with respect to cut-off values used and the number of DNA mutations included in the screening strategies was observed, and the methodological quality differed considerably between studies.

Conclusions

The evidence suggested that (i) all screening strategies are cost-effective as compared to the no-screening option and (ii) IRT-PAP seems to be the most cost-effective screening strategy towards CFNBS. Methodological and contextual differences of the individual studies make it difficult to derive strong conclusions from this evidence. Nevertheless, from a health-economic perspective, IRT-PAP should be included as an alternative when deciding on the screening strategy in the implementation of CFNBS.