STUDY OBJECTIVE: Evaluating the costs, effects, and savings of several strategies for cystic fibrosis (CF) gene carrier screening. DESIGN: A general model for evaluating prenatal, preconceptional, school, and neonatal carrier screening was constructed. For prenatal and preconceptional screening, two strategies were evaluated: single entry and double entry two step couple screening. Firstly, the Dutch situation was evaluated prospectively; subsequently the results were generalised to other carrier frequencies. SETTING: Prospective simulation model. MAIN RESULTS: Of all screening strategies, neonatal carrier screening gives most carrier couples an informed choice concerning reproduction. If the parents of carrier newborns would not be tested however, prenatal screening detects most carrier couples. Prenatal and single entry preconceptional screening programmes have a favourable cost-savings balance in the Netherlands under a wide range of assumptions. For double entry preconceptional screening and neonatal screening, high enough values of uptake of screening, prenatal diagnosis, and induced abortion are necessary. School carrier screening does not have a favourable cost-savings balance. CONCLUSIONS: If a CF screening programme is judged to be useful on individual and social grounds, costs considerations are no obstacle for prenatal and single entry preconceptional screening.

*Decision Support Techniques, *Models, Economic, Child, Cost-Benefit Analysis, Costs and Cost Analysis, Cystic Fibrosis/genetics/*prevention & control, Female, Genetic Counseling/economics, Genetic Screening/*economics/methods, Heterozygote Detection, Humans, Infant, Newborn, Male, Prenatal Diagnosis/economics, Prospective Studies, Research Support, Non-U.S. Gov't
Journal of Epidemiology and Community Health
Erasmus MC: University Medical Center Rotterdam

Wildhagen, M.F, Hilderink, H.B, Verzijl, J.G, Verheij, J.B, Kooij, L, Tijmstra, T, … Habbema, J.D.F. (1998). Costs, effects, and savings of screening for cystic fibrosis gene carriers. Journal of Epidemiology and Community Health. Retrieved from