Telomerase mutations in smokers with severe emphysema.

نویسندگان

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

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.

برای دانلود متن کامل این مقاله و بیش از 32 میلیون مقاله دیگر ابتدا ثبت نام کنید

ثبت نام

اگر عضو سایت هستید لطفا وارد حساب کاربری خود شوید

منابع مشابه

Emphysema Predicts Hospitalisation and Incident Airflow Obstruction among Older Smokers: A Prospective Cohort Study

BACKGROUND Emphysema on CT is common in older smokers. We hypothesised that emphysema on CT predicts acute episodes of care for chronic lower respiratory disease among older smokers. MATERIALS AND METHODS Participants in a lung cancer screening study age ≥ 60 years were recruited into a prospective cohort study in 2001-02. Two radiologists independently visually assessed the severity of emphy...

متن کامل

Telomerase reverse-transcriptase homozygous mutations in autosomal recessive dyskeratosis congenita and Hoyeraal-Hreidarsson syndrome.

Dyskeratosis congenita (DC) is a multisystem bone marrow failure syndrome characterized by a triad of mucocutaneous abnormalities and an increased predisposition to malignancy. X-linked DC is due to mutations in DKC1, while heterozygous mutations in TERC (telomerase RNA component) and TERT (telomerase reverse transcriptase) have been found in autosomal dominant DC. Many patients with DC remain ...

متن کامل

CHRONIC OBSTRUCTIVE PULMONARY DISEASE Decreased expression of interleukin 13 in human lung emphysema

Background: The overexpression of interferon (IFN)c or interleukin (IL)-13 in the adult murine lung induces the development of changes that mirror human lung emphysema. Methods: IL-13 and IFNc expression was determined in lung samples from five groups of patients: severe emphysema without a1-antitrypsin deficiency (SE+, n = 10); severe emphysema with a1-antitrypsin deficiency (SE2, n = 5); mild...

متن کامل

Morphological and cellular basis for airflow limitation in smokers.

Airflow limitation has two well-defined components, increased resistance, which is found predominantly in the small airways, and loss of elastic recoil. Small airways contribute to the increased resistance to flow by the narrowing of the airway lumen. Morphometric studies have shown that smokers have increased epithelial abnormalities, cellular inflammatory infiltrates in the airway wall, incre...

متن کامل

Decreased expression of interleukin 13 in human lung emphysema.

BACKGROUND The overexpression of interferon (IFN)gamma or interleukin (IL)-13 in the adult murine lung induces the development of changes that mirror human lung emphysema. METHODS IL-13 and IFNgamma expression was determined in lung samples from five groups of PATIENTS severe emphysema without alpha(1)-antitrypsin deficiency (SE+, n = 10); severe emphysema with alpha(1)-antitrypsin deficien...

متن کامل

ذخیره در منابع من


  با ذخیره ی این منبع در منابع من، دسترسی به آن را برای استفاده های بعدی آسان تر کنید

برای دانلود متن کامل این مقاله و بیش از 32 میلیون مقاله دیگر ابتدا ثبت نام کنید

ثبت نام

اگر عضو سایت هستید لطفا وارد حساب کاربری خود شوید

عنوان ژورنال:
  • The Journal of clinical investigation

دوره 125 2  شماره 

صفحات  -

تاریخ انتشار 2015