Newborn screening for cystic fibrosis is associated with the lowest
healthcare costs : a 10-year observational follow-up study in France
Abstract
Objectives: This study aims to study the healthcare (HC) costs
associated with cystic fibrosis (CF) in children diagnosed prenatally
(ANT), through newborn screening (NBS), after birth due to meconium
ileus (MI), or later based on symptoms (LS). Additionally, it seeks to
clinically characterize children with CF (chCF) with different
trajectories of HC costs. Study design: A retrospective
observational study was conducted on data from the French CF Registry
(FCFR) and the French National Claims Database (SNDS) linked from 2006
to 2021. HC costs related to CF diagnosis circumstances were estimated
per year of life among chCF up to age 10. Group-based trajectory
modeling was performed to identify subgroups with similar cost
trajectories. Results: Between 2006 and 2011, data from 1,065
chCF were recorded in the FCFR. 973 (91.4%) were matched with SNDS, and
779 (73.1%) had at least 10 years of follow-up. NBS resulted in the
lowest costs. During the first year, HC costs of chCF diagnosed with MI
and ANT were higher than for those diagnosed with NBS or LS. Expenses
declined in the second year of life and then gradually increased to
approximately €20,000 by the tenth year. Three groups with different
cost trajectories were identified. Groups with the highest costs had a
lower lung function at 6 and 10 years and the lowest weight and height
z-scores at 2 and 10 years (all p<0.05). Conclusion: HC
costs were lower in chCF diagnosed by NBS, and high HC costs could occur
at the earliest stages of CF.