Onset of persistent pseudomonas aeruginosa infection in children with cystic fibrosis with interval censored data.

TitleOnset of persistent pseudomonas aeruginosa infection in children with cystic fibrosis with interval censored data.
Publication TypeJournal Article
Year of Publication2016
AuthorsWang, Wenjie, Ming-Hui Chen, Sy Han Chiou, Hui-Chuan Lai, Xiaojing Wang, Jun Yan, and Zhumin Zhang
JournalBMC Med Res Methodol
Volume16
Issue1
Pagination122
Date Published2016 09 17
ISSN1471-2288
KeywordsAlgorithms, Child, Preschool, Cystic Fibrosis, Early Diagnosis, Female, Humans, Infant, Infant, Newborn, Male, Markov Chains, Monte Carlo Method, Neonatal Screening, Proportional Hazards Models, Pseudomonas aeruginosa, Pseudomonas Infections, Risk
Abstract

BACKGROUND: Persistent Pseudomonas aeruginosa (PPA) infection promotes lung function deterioration in children with cystic fibrosis (CF). Although early CF diagnosis through newborn screening (NBS) has been shown to provide nutritional/growth benefit, it is unclear whether NBS lowers the risk of PPA infection and how the effect of NBS vary with age. Modeling the onset age of PPA infection is challenging because 1) the onset age of PPA infection is interval censored in patient registry data; and 2) some risk factors such as NBS may have time-varying effects.METHODS: This problem fits into the framework of a recently developed Bayesian dynamic Cox model for interval censored data, where each regression coefficient is allowed to be time-varying to an extent determined by the data.RESULTS: Application of the methodology to data from the CF Foundation Patient Registry revealed interesting findings. Compared with patients with meconium ileus or diagnosed through signs or symptoms, patients diagnosed through NBS had significantly lower risks of acquiring PPA infection between age 1 and 2 years, and the benefit in survival rate was found to last up to age 4 years. Two cohorts of five years apart were compared. Patients born in cohort 2003-2004 had significantly lower risks of the PPA infections at any age up to 4 years than those born in 1998-1999.CONCLUSIONS: The study supports benefits of NBS on PPA infection in early childhood. In addition, our analyses demonstrate that patients in the more recent cohort had significantly lower risks of acquiring PPA infection up to age 4 years, which suggests improved CF treatment and care over time.

DOI10.1186/s12874-016-0220-5
Alternate JournalBMC Med Res Methodol
Original PublicationOnset of persistent pseudomonas aeruginosa infection in children with cystic fibrosis with interval censored data.
PubMed ID27639560
PubMed Central IDPMC5027124
Grant ListT32 NS048005 / NS / NINDS NIH HHS / United States
R01 GM070335 / GM / NIGMS NIH HHS / United States
R01 DK109692 / DK / NIDDK NIH HHS / United States
R01 DK072126 / DK / NIDDK NIH HHS / United States
P01 CA142538 / CA / NCI NIH HHS / United States