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

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...

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Autores principales: Wang, Wenjie, Chen, Ming-Hui, Chiou, Sy Han, Lai, Hui-Chuan, Wang, Xiaojing, Yan, Jun, Zhang, Zhumin
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2016
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5027124/
https://www.ncbi.nlm.nih.gov/pubmed/27639560
http://dx.doi.org/10.1186/s12874-016-0220-5
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author Wang, Wenjie
Chen, Ming-Hui
Chiou, Sy Han
Lai, Hui-Chuan
Wang, Xiaojing
Yan, Jun
Zhang, Zhumin
author_facet Wang, Wenjie
Chen, Ming-Hui
Chiou, Sy Han
Lai, Hui-Chuan
Wang, Xiaojing
Yan, Jun
Zhang, Zhumin
author_sort Wang, Wenjie
collection PubMed
description 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. ELECTRONIC SUPPLEMENTARY MATERIAL: The online version of this article (doi:10.1186/s12874-016-0220-5) contains supplementary material, which is available to authorized users.
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spelling pubmed-50271242016-09-22 Onset of persistent pseudomonas aeruginosa infection in children with cystic fibrosis with interval censored data Wang, Wenjie Chen, Ming-Hui Chiou, Sy Han Lai, Hui-Chuan Wang, Xiaojing Yan, Jun Zhang, Zhumin BMC Med Res Methodol Research Article 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. ELECTRONIC SUPPLEMENTARY MATERIAL: The online version of this article (doi:10.1186/s12874-016-0220-5) contains supplementary material, which is available to authorized users. BioMed Central 2016-09-17 /pmc/articles/PMC5027124/ /pubmed/27639560 http://dx.doi.org/10.1186/s12874-016-0220-5 Text en © The Author(s) 2016 Open Access This article is distributed under the terms of the Creative Commons Attribution 4.0 International License(http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver(http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
spellingShingle Research Article
Wang, Wenjie
Chen, Ming-Hui
Chiou, Sy Han
Lai, Hui-Chuan
Wang, Xiaojing
Yan, Jun
Zhang, Zhumin
Onset of persistent pseudomonas aeruginosa infection in children with cystic fibrosis with interval censored data
title Onset of persistent pseudomonas aeruginosa infection in children with cystic fibrosis with interval censored data
title_full Onset of persistent pseudomonas aeruginosa infection in children with cystic fibrosis with interval censored data
title_fullStr Onset of persistent pseudomonas aeruginosa infection in children with cystic fibrosis with interval censored data
title_full_unstemmed Onset of persistent pseudomonas aeruginosa infection in children with cystic fibrosis with interval censored data
title_short Onset of persistent pseudomonas aeruginosa infection in children with cystic fibrosis with interval censored data
title_sort onset of persistent pseudomonas aeruginosa infection in children with cystic fibrosis with interval censored data
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5027124/
https://www.ncbi.nlm.nih.gov/pubmed/27639560
http://dx.doi.org/10.1186/s12874-016-0220-5
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