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Published in: BMC Medical Research Methodology 1/2016

Open Access 01-12-2016 | Research Article

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

Authors: Wenjie Wang, Ming-Hui Chen, Sy Han Chiou, Hui-Chuan Lai, Xiaojing Wang, Jun Yan, Zhumin Zhang

Published in: BMC Medical Research Methodology | Issue 1/2016

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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.
Appendix
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Metadata
Title
Onset of persistent pseudomonas aeruginosa infection in children with cystic fibrosis with interval censored data
Authors
Wenjie Wang
Ming-Hui Chen
Sy Han Chiou
Hui-Chuan Lai
Xiaojing Wang
Jun Yan
Zhumin Zhang
Publication date
01-12-2016
Publisher
BioMed Central
Published in
BMC Medical Research Methodology / Issue 1/2016
Electronic ISSN: 1471-2288
DOI
https://doi.org/10.1186/s12874-016-0220-5

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