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Whole exome sequencing identifies novel candidate genes that modify chronic obstructive pulmonary disease susceptibility
Journal article   Open access   Peer reviewed

Whole exome sequencing identifies novel candidate genes that modify chronic obstructive pulmonary disease susceptibility

Shannon Bruse, Michael Moreau, Yana Bromberg, Jun-Ho Jang, Nan Wang, Hongseok Ha, Maria Picchi, Yong Lin, Raymond J Langley, Clifford Qualls, …
Human genomics, Vol.10(1), pp.1-1
01/07/2016
DOI: 10.1186/s40246-015-0058-7
PMCID: PMC4705629
PMID: 26744305
url
https://doi.org/10.1186/s40246-015-0058-7View
Published (Version of record) Open Access

Abstract

Chronic obstructive pulmonary disease (COPD) is characterized by an irreversible airflow limitation in response to inhalation of noxious stimuli, such as cigarette smoke. However, only 15-20 % smokers manifest COPD, suggesting a role for genetic predisposition. Although genome-wide association studies have identified common genetic variants that are associated with susceptibility to COPD, effect sizes of the identified variants are modest, as is the total heritability accounted for by these variants. In this study, an extreme phenotype exome sequencing study was combined with in vitro modeling to identify COPD candidate genes. We performed whole exome sequencing of 62 highly susceptible smokers and 30 exceptionally resistant smokers to identify rare variants that may contribute to disease risk or resistance to COPD. This was a cross-sectional case-control study without therapeutic intervention or longitudinal follow-up information. We identified candidate genes based on rare variant analyses and evaluated exonic variants to pinpoint individual genes whose function was computationally established to be significantly different between susceptible and resistant smokers. Top scoring candidate genes from these analyses were further filtered by requiring that each gene be expressed in human bronchial epithelial cells (HBECs). A total of 81 candidate genes were thus selected for in vitro functional testing in cigarette smoke extract (CSE)-exposed HBECs. Using small interfering RNA (siRNA)-mediated gene silencing experiments, we showed that silencing of several candidate genes augmented CSE-induced cytotoxicity in vitro. Our integrative analysis through both genetic and functional approaches identified two candidate genes (TACC2 and MYO1E) that augment cigarette smoke (CS)-induced cytotoxicity and, potentially, COPD susceptibility.
Phenotype Smoking - adverse effects Genetic Predisposition to Disease Humans Middle Aged Cell Survival - genetics Genetic Association Studies - methods Male Smoking - genetics Pulmonary Disease, Chronic Obstructive - pathology Exome - genetics Female Aged Primary Cell Culture High-Throughput Nucleotide Sequencing - methods Pulmonary Disease, Chronic Obstructive - genetics

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