Kelley T J, Al-Nakkash L, Cotton C U, Drumm M L
Department of Pediatrics, Case Western Reserve University, Cleveland, Ohio 44106-4948, USA.
J Clin Invest. 1996 Jul 15;98(2):513-20. doi: 10.1172/JCI118819.
Many heterologously expressed mutants of the cystic fibrosis transmembrane conductance regulator (CFTR) exhibit residual chloride channel activity that can be stimulated by agonists of the adenylate cyclase/protein kinase A pathway. Because of clinical implications for cystic fibrosis of activating mutants in vivo, we are investigating whether deltaF508, the most common disease-associated CFTR mutation, can be activated in airway epithelial cells. We have found that, 36Cl- efflux can be stimulated 19-61% above baseline by beta-adrenoreceptor agonists and cGI-phosphodiesterase inhibitors in transformed nasal polyp (CF-T43) cells homozygous for the deltaF508 mutation. The increase in 36Cl- permeability is diminished by protein kinase A inhibitors and is not mediated by an increase in intracellular calcium concentrations. Preincubation of CF-T43 cells with CFTR anti-sense oligonucleotides prevented an increase in 36Cl- efflux in response to beta-agonist and phosphodiesterase inhibitor. Primary cells isolated from CF nasal polyps gave similar results. These data indicate that endogenous levels of deltaF508 protein can be stimulated to increase 36Cl- permeability in airway epithelial cells.
许多囊性纤维化跨膜传导调节因子(CFTR)的异源表达突变体表现出残余的氯离子通道活性,该活性可被腺苷酸环化酶/蛋白激酶A途径的激动剂所刺激。鉴于激活突变体在体内对囊性纤维化的临床意义,我们正在研究最常见的与疾病相关的CFTR突变ΔF508是否能在气道上皮细胞中被激活。我们发现,在纯合ΔF508突变的转化鼻息肉(CF-T43)细胞中,β-肾上腺素能受体激动剂和环鸟苷酸磷酸二酯酶抑制剂可使³⁶Cl⁻外流比基线水平增加19% - 61%。³⁶Cl⁻通透性的增加被蛋白激酶A抑制剂减弱,且不是由细胞内钙浓度的增加介导的。用CFTR反义寡核苷酸预孵育CF-T43细胞可阻止其对β-激动剂和磷酸二酯酶抑制剂的³⁶Cl⁻外流增加。从CF鼻息肉中分离的原代细胞也得到了类似的结果。这些数据表明,内源性水平的ΔF508蛋白可被刺激以增加气道上皮细胞中的³⁶Cl⁻通透性。