Department of Medicine, Roy J. and Lucille A. Carver College of Medicine, University of Iowa, Iowa City, Iowa 52242, USA.
Nature. 2012 Jul 4;487(7405):109-13. doi: 10.1038/nature11130.
Cystic fibrosis (CF) is a life-shortening disease caused by mutations in the cystic fibrosis transmembrane conductance regulator (CFTR) gene. Although bacterial lung infection and the resulting inflammation cause most of the morbidity and mortality, how the loss of CFTR function first disrupts airway host defence has remained uncertain. To investigate the abnormalities that impair elimination when a bacterium lands on the pristine surface of a newborn CF airway, we interrogated the viability of individual bacteria immobilized on solid grids and placed onto the airway surface. As a model, we studied CF pigs, which spontaneously develop hallmark features of CF lung disease. At birth, their lungs lack infection and inflammation, but have a reduced ability to eradicate bacteria. Here we show that in newborn wild-type pigs, the thin layer of airway surface liquid (ASL) rapidly kills bacteria in vivo, when removed from the lung and in primary epithelial cultures. Lack of CFTR reduces bacterial killing. We found that the ASL pH was more acidic in CF pigs, and reducing pH inhibited the antimicrobial activity of ASL. Reducing ASL pH diminished bacterial killing in wild-type pigs, and, conversely, increasing ASL pH rescued killing in CF pigs. These results directly link the initial host defence defect to the loss of CFTR, an anion channel that facilitates HCO(3)(-) transport. Without CFTR, airway epithelial HCO(3)(-) secretion is defective, the ASL pH falls and inhibits antimicrobial function, and thereby impairs the killing of bacteria that enter the newborn lung. These findings suggest that increasing ASL pH might prevent the initial infection in patients with CF, and that assaying bacterial killing could report on the benefit of therapeutic interventions.
囊性纤维化 (CF) 是一种由囊性纤维化跨膜电导调节因子 (CFTR) 基因突变引起的缩短寿命的疾病。尽管细菌肺部感染和由此产生的炎症导致了大部分发病率和死亡率,但 CFTR 功能丧失如何首先破坏气道宿主防御仍然不确定。为了研究当细菌降落在新生 CF 气道的原始表面时,哪些异常会削弱清除作用,我们研究了固定在固体网格上并放置在气道表面上的单个细菌的存活能力。作为一个模型,我们研究了 CF 猪,它们会自发地发展出 CF 肺部疾病的标志性特征。出生时,它们的肺部没有感染和炎症,但清除细菌的能力降低。在这里,我们表明,在新生的野生型猪中,当从肺部和原代上皮培养物中取出时,气道表面液体 (ASL) 的薄层会迅速杀死体内的细菌。CFTR 的缺乏会降低细菌的杀伤能力。我们发现 CF 猪的 ASL pH 值更酸性,并且降低 pH 值会抑制 ASL 的抗菌活性。降低 ASL pH 值会减弱野生型猪的细菌杀伤能力,反之亦然,增加 ASL pH 值会挽救 CF 猪的杀伤能力。这些结果直接将初始宿主防御缺陷与 CFTR 的丧失联系起来,CFTR 是一种促进 HCO3(-) 转运的阴离子通道。没有 CFTR,气道上皮的 HCO3(-) 分泌功能缺陷,ASL pH 值下降并抑制抗菌功能,从而削弱了进入新生肺部的细菌的杀伤能力。这些发现表明,增加 ASL pH 值可能会预防 CF 患者的初始感染,并且测定细菌杀伤能力可以报告治疗干预的益处。
Am J Physiol Lung Cell Mol Physiol. 2016-4-1
Eur J Cell Biol. 2018-6-18
Am J Physiol Lung Cell Mol Physiol. 2024-11-1
Proc Natl Acad Sci U S A. 2014-9-2
NPJ Biofilms Microbiomes. 2025-6-12
Antioxidants (Basel). 2025-3-4
Pediatr Nephrol. 2025-3-17
Mol Ther Methods Clin Dev. 2024-6-24
Am J Physiol Lung Cell Mol Physiol. 2025-2-1
Am J Physiol Cell Physiol. 2010-10-6
Sci Transl Med. 2010-4-28