Strong T V, Wilkinson D J, Mansoura M K, Devor D C, Henze K, Yang Y, Wilson J M, Cohn J A, Dawson D C, Frizzell R A
Department of Human Genetics, Howard Hughes Medical Institute, University of Michigan, Ann Arbor 48109.
Hum Mol Genet. 1993 Mar;2(3):225-30. doi: 10.1093/hmg/2.3.225.
The cystic fibrosis transmembrane conductance regulator (CFTR) gene encodes a cAMP-activated chloride (Cl-) channel, and expression of the full length gene in vitro is sufficient to correct the Cl- conductance defect that is characteristic of cystic fibrosis (CF) epithelial cells. Alternatively spliced forms of CFTR mRNA have been identified in several tissues from normal individuals. One of the alternative transcripts, often present at high levels, results in the in-frame deletion of exon 9. Translation of this transcript would result in a CFTR protein missing the amino terminal portion of the first nucleotide binding fold (NBF). To evaluate the possible function of this form of CFTR, a cDNA representing this transcript (CFTR delta 9) was transduced into CFPAC cells, which are derived from a CF patient. CFTR delta 9 RNA was expressed in the transduced cell lines, but only immature, incompletely glycosylated protein was detectable by Western blot analysis. No increase in cAMP-activated anion permeability was detectable by 125I efflux assay or by means of the halide sensitive dye 6-methoxy-N-(3-sulfopropyl) quinolinium (SPQ). In a second assay system, in vitro synthesized mRNA representing CFTR delta D9 was injected into Xenopus oocytes, but expression of this alternatively spliced form of CFTR was not associated with the appearance of Cl- conductance. These results suggest that the protein produced by the CFTR delta 9 transcript is not properly processed and is not capable of generating Cl- conductance in response to cAMP. Whether this alternative transcript has some other function or represents 'noise' in the mRNA splicing mechanism remains unresolved.
囊性纤维化跨膜传导调节因子(CFTR)基因编码一种cAMP激活的氯离子(Cl-)通道,全长基因在体外的表达足以纠正囊性纤维化(CF)上皮细胞特有的Cl-传导缺陷。在正常个体的多个组织中已鉴定出CFTR mRNA的可变剪接形式。其中一种可变转录本,通常高水平存在,导致外显子9的框内缺失。该转录本的翻译将产生一种缺少第一个核苷酸结合结构域(NBF)氨基末端部分的CFTR蛋白。为了评估这种形式的CFTR的可能功能,将代表该转录本的cDNA(CFTR delta 9)转导到源自CF患者的CFPAC细胞中。CFTR delta 9 RNA在转导的细胞系中表达,但通过蛋白质印迹分析仅可检测到未成熟、不完全糖基化的蛋白质。通过125I流出试验或卤化物敏感染料6-甲氧基-N-(3-磺丙基)喹啉鎓(SPQ)均未检测到cAMP激活的阴离子通透性增加。在第二个试验系统中,将代表CFTR delta D9的体外合成mRNA注射到非洲爪蟾卵母细胞中,但这种CFTR可变剪接形式的表达与Cl-传导的出现无关。这些结果表明,CFTR delta 9转录本产生的蛋白质没有得到正确加工,并且不能响应cAMP产生Cl-传导。这种可变转录本是否具有其他功能或代表mRNA剪接机制中的“噪音”仍未解决。