Original articlePotential impact of newborn screening for cystic fibrosis on child survival: A systematic review and analysis
Section snippets
Methods
In preparation for the 2003 workshop, the scientific literature from 1997 through July 2003 was reviewed via a search of Medline for articles with abstracts addressing “newborn screening” and “cystic fibrosis” published in English or French. A total of five studies that investigated outcomes in screened cohorts or mixed cohorts of screened and unscreened children with CF were identified.12, 13, 14, 15, 16 Subsequent searches of Medline yielded one additional publication of original data.17 In
Review
The strongest evidence for causality in terms of health outcomes of screening comes from randomized controlled trials (RCTs). In the United Kingdom, all babies born in Wales and the West Midlands during 1985 to 1989 were randomly allocated to undergo or not undergo CF screening on an alternate-week basis.22 The study did not have a standard treatment or follow-up protocol, and ascertainment in the unscreened group was incomplete. Investigators subsequently reviewed registry and death
Discussion
CF NBS has not been considered by policy makers as a life-saving intervention, unlike screening for certain disorders, such as sickle cell disease (SCD) or galactosemia.28 Previous reviewers have expressed skepticism that NBS for CF is likely to reduce CF-related mortality.2, 3 The CDC report noted that several studies had found lower mortality in screened cohorts, but it raised questions about statistical significance and potential publication bias.11 On the other hand, the finding of improved
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