Int J Technol Assess Health Care. 2018 Jan;34(2):189-195. doi: 10.1017/S0266462318000181. Epub 2018 Apr 10.


Objectives: This paper aims to describe the added value of combining cost-effectiveness and ethical evaluations when the preferences of the decision maker toward cost-effectiveness evaluation outcomes are not known, with the French national neonatal screening of cystic fibrosis (CF) as a case-study.

Methods: A cost-effectiveness analysis comparing four CF neonatal screening strategies, with or without DNA testing, was performed. Ethical positions toward their outcomes were described. In addition, a post-hoc analysis of the ethical issues being considered relevant from the decision-makers' perspective was conducted.

Results: Two strategies were found equally cost-effective. Among them, choosing the non-DNA or a DNA-based strategy constrains the decision maker to render a judgement between different ethical issues or disagreements associated with the screening program.

Conclusions: The analysis supports the relevance of combining cost-effectiveness and ethics evaluation in developing health policy, as a way to reveal or clarify the motives associated with health. The choice of the decision maker to favor the DNA-based strategy, which was not originally recommended, creates the opportunity to make explicit the role played by ethical issues in the decision.

Keywords: Cost-effectiveness; Ethics; Screening.

MeSH terms

  • Cost-Benefit Analysis
  • Cystic Fibrosis / diagnosis*
  • Cystic Fibrosis / genetics
  • Decision Making*
  • Diagnostic Errors
  • France
  • Genetic Testing
  • Humans
  • Infant, Newborn
  • Neonatal Screening / economics*
  • Neonatal Screening / ethics*
  • Pancreatitis-Associated Proteins / blood
  • Trypsinogen / blood
  • Uncertainty


  • Pancreatitis-Associated Proteins
  • Trypsinogen