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Telomerase mutations in smokers with severe emphysema.

Authors :
Stanley SE
Chen JJ
Podlevsky JD
Alder JK
Hansel NN
Mathias RA
Qi X
Rafaels NM
Wise RA
Silverman EK
Barnes KC
Armanios M
Source :
The Journal of clinical investigation [J Clin Invest] 2015 Feb; Vol. 125 (2), pp. 563-70. Date of Electronic Publication: 2014 Dec 22.
Publication Year :
2015

Abstract

Mutations in the essential telomerase genes TERT and TR cause familial pulmonary fibrosis; however, in telomerase-null mice, short telomeres predispose to emphysema after chronic cigarette smoke exposure. Here, we tested whether telomerase mutations are a risk factor for human emphysema by examining their frequency in smokers with chronic obstructive pulmonary disease (COPD). Across two independent cohorts, we found 3 of 292 severe COPD cases carried deleterious mutations in TERT (1%). This prevalence is comparable to the frequency of alpha-1 antitrypsin deficiency documented in this population. The TERT mutations compromised telomerase catalytic activity, and mutation carriers had short telomeres. Telomerase mutation carriers with emphysema were predominantly female and had an increased incidence of pneumothorax. In families, emphysema showed an autosomal dominant inheritance pattern, along with pulmonary fibrosis and other telomere syndrome features, but manifested only in smokers. Our findings identify germline mutations in telomerase as a Mendelian risk factor for COPD susceptibility that clusters in autosomal dominant families with telomere-mediated disease including pulmonary fibrosis.

Details

Language :
English
ISSN :
1558-8238
Volume :
125
Issue :
2
Database :
MEDLINE
Journal :
The Journal of clinical investigation
Publication Type :
Academic Journal
Accession number :
25562321
Full Text :
https://doi.org/10.1172/JCI78554