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.

Originele taal-2English
Pagina's (van-tot)306-315
Aantal pagina's10
TijdschriftJournal of Cystic Fibrosis
Nummer van het tijdschrift3
Vroegere onlinedatum20 mrt 2018
StatusPublished - mei 2018


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