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特发性肺纤维化:遗传发现的最新进展。

Idiopathic pulmonary fibrosis: update on genetic discoveries.

机构信息

University of Texas Southwestern Medical Center, Dallas, TX 75390-8591, USA.

出版信息

Proc Am Thorac Soc. 2011 May;8(2):158-62. doi: 10.1513/pats.201008-056MS.

Abstract

Idiopathic pulmonary fibrosis (IPF) is a progressive fibrotic disease of the lungs that increases in prevalence with advanced age. Recent evidence indicates that mutations in genes of two different biologic pathways lead to the common phenotype of familial pulmonary fibrosis (FPF) and sporadic IPF. Mutations in the genes encoding the lung surfactant proteins C and A2 (SFTPC and SFTPA2, respectively) cause increased endoplasmic reticulum stress in type II alveolar epithelial cells. Mutations in the genes encoding telomerase (TERT and TERC) cause IPF through shortening of telomere lengths and probable exhaustion of lung stem cells. All of the mutations are individually rare, but, collectively, TERT mutations are the most common genetic defect found in FPF. The overall penetrance of pulmonary fibrosis in TERT mutation carriers is 40% in subjects with a mean age of 51 years. Penetrance increases with advanced age, is greater in males than in females, and is positively associated with fibrogenic environmental exposures. Short telomere lengths are found in patients with FPF and sporadic IPF without mutations in telomerase, suggesting that the biologic pathway of telomerase dysfunction provides a biologic explanation for the age-related prevalence of IPF. The molecular data of two seemingly unrelated biologic pathways-alveolar epithelial endoplasmic reticulum stress and telomerase dysfunction-are beginning to elucidate the pathogenesis of IPF. These results have potentially predictive and therapeutic value.

摘要

特发性肺纤维化(IPF)是一种进行性肺纤维化疾病,随着年龄的增长而患病率增加。最近的证据表明,两种不同生物学途径的基因突变导致家族性肺纤维化(FPF)和散发性 IPF 的常见表型。编码肺表面活性剂蛋白 C 和 A2(分别为 SFTPC 和 SFTPA2)的基因突变导致 II 型肺泡上皮细胞内质网应激增加。编码端粒酶(TERT 和 TERC)的基因突变通过缩短端粒长度和可能耗尽肺干细胞导致 IPF。所有突变都是单独罕见的,但 TERT 突变是 FPF 中最常见的遗传缺陷。在平均年龄为 51 岁的 TERT 突变携带者中,肺纤维化的总外显率为 40%。外显率随年龄增长而增加,男性大于女性,与纤维形成性环境暴露呈正相关。在没有端粒酶基因突变的 FPF 和散发性 IPF 患者中发现端粒较短,这表明端粒酶功能障碍的生物学途径为 IPF 的年龄相关性患病率提供了生物学解释。两种看似无关的生物学途径——肺泡上皮内质网应激和端粒酶功能障碍的分子数据——开始阐明 IPF 的发病机制。这些结果具有潜在的预测和治疗价值。

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本文引用的文献

1
Telomere lengths, pulmonary fibrosis and telomerase (TERT) mutations.
PLoS One. 2010 May 19;5(5):e10680. doi: 10.1371/journal.pone.0010680.
2
Surfactant protein A2 mutations associated with pulmonary fibrosis lead to protein instability and endoplasmic reticulum stress.
J Biol Chem. 2010 Jul 16;285(29):22103-13. doi: 10.1074/jbc.M110.121467. Epub 2010 May 13.
3
Telomere diseases.
N Engl J Med. 2009 Dec 10;361(24):2353-65. doi: 10.1056/NEJMra0903373.
4
Genetic defects in surfactant protein A2 are associated with pulmonary fibrosis and lung cancer.
Am J Hum Genet. 2009 Jan;84(1):52-9. doi: 10.1016/j.ajhg.2008.11.010. Epub 2008 Dec 18.
6
Short telomeres are a risk factor for idiopathic pulmonary fibrosis.
Proc Natl Acad Sci U S A. 2008 Sep 2;105(35):13051-6. doi: 10.1073/pnas.0804280105. Epub 2008 Aug 27.
7
Epithelial endoplasmic reticulum stress and apoptosis in sporadic idiopathic pulmonary fibrosis.
Am J Respir Crit Care Med. 2008 Oct 15;178(8):838-46. doi: 10.1164/rccm.200802-313OC. Epub 2008 Jul 17.
8
Telomere shortening in familial and sporadic pulmonary fibrosis.
Am J Respir Crit Care Med. 2008 Oct 1;178(7):729-37. doi: 10.1164/rccm.200804-550OC. Epub 2008 Jul 17.
9
Human diseases of telomerase dysfunction: insights into tissue aging.
Nucleic Acids Res. 2007;35(22):7406-16. doi: 10.1093/nar/gkm644. Epub 2007 Oct 2.
10
Mortality from pulmonary fibrosis increased in the United States from 1992 to 2003.
Am J Respir Crit Care Med. 2007 Aug 1;176(3):277-84. doi: 10.1164/rccm.200701-044OC. Epub 2007 May 3.

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