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DOI 10.1172/jci78554

Медицинская библиотека · The Journal of clinical investigation · 2014

Telomerase mutations in smokers with severe emphysema.

Stanley Susan E, Chen Julian J L, Podlevsky Joshua D, Alder Jonathan K, Hansel Nadia N, Mathias Rasika A, Qi Xiaodong, Rafaels Nicholas M, Wise Robert A, Silverman Edwin K, Barnes Kathleen C, Armanios Mary

Клиническое исследованиеСтатьяПоддержка NIH: внешняя программаПеревод пока не подготовлен

Аннотация

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.

Для цитирования: Stanley Susan E, Chen Julian J L, Podlevsky Joshua D, Alder Jonathan K, Hansel Nadia N, Mathias Rasika A, Qi Xiaodong, Rafaels Nicholas M, Wise Robert A, Silverman Edwin K, Barnes Kathleen C, Armanios Mary. Telomerase mutations in smokers with severe emphysema.. The Journal of clinical investigation. 2014. DOI: 10.1172/jci78554.

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