University of Texas Southwestern Medical Center, Dallas, TX 75390-8591, USA.
Proc Am Thorac Soc. 2011 May;8(2):158-62. doi: 10.1513/pats.201008-056MS.
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.
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。