Economic evaluation of cystic fibrosis screening: a review of the literature.

Objectives: To critically examine the economic evidence regarding cystic fibrosis (CF) carrier screening and to understand issues relating to the transferability of international findings to any national context for policy decisions.

Methods: A systematic literature search identified 14 studies (out of 29 economic studies on CF) focusing on preconception or prenatal screening between 1990 and 2006. These studies were then assessed against international benchmarks on conducting and reporting of economic evaluations, costing methodology used and focusing on the transferability of the evidence to national contexts.

Results: The primary outcome measures varied considerably between studies and there was considerable ambiguity and variation on how costs were estimated. The Incremental Cost Effectiveness Ratio (ICER) and net savings, for preconception and prenatal screening were inconsistent and varied significantly, even after adjusting for timing and exchange rates. Differences in screening participation rates, reproductive choices, test sensitivity, cost of test and lifetime cost of care make up a large part of the ICER variations.

Conclusion: The heterogeneity in study design, model inputs and reporting of economic evaluations of CF carrier screening makes comparability and transferability across countries and even within countries difficult. This reinforces the need to assess any technology within the relevant context, and to not simply generalize from reported studies. In turn, this adds to the complex task of making efficient resource allocation decisions in the area of CF carrier screening. Our evaluation adds weight to the calls for revisiting the way economic studies are conducted and reported.