Literature DB >> 7684641

Expression of an abundant alternatively spliced form of the cystic fibrosis transmembrane conductance regulator (CFTR) gene is not associated with a cAMP-activated chloride conductance.

T V Strong1, D J Wilkinson, M K Mansoura, D C Devor, K Henze, Y Yang, J M Wilson, J A Cohn, D C Dawson, R A Frizzell.   

Abstract

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.

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Year:  1993        PMID: 7684641     DOI: 10.1093/hmg/2.3.225

Source DB:  PubMed          Journal:  Hum Mol Genet        ISSN: 0964-6906            Impact factor:   6.150


  20 in total

1.  Whole-gene CFTR sequencing combined with digital RT-PCR improves genetic diagnosis of cystic fibrosis.

Authors:  Letizia Straniero; Giulia Soldà; Lucy Costantino; Manuela Seia; Paola Melotti; Carla Colombo; Rosanna Asselta; Stefano Duga
Journal:  J Hum Genet       Date:  2016-08-04       Impact factor: 3.172

2.  Association of cystic fibrosis transmembrane conductance regulator gene variants with acute lung injury in African American children with pneumonia*.

Authors:  Julie M Baughn; Michael W Quasney; Pippa Simpson; Daniel Merchant; Shun-Hwa Li; Hara Levy; Mary K Dahmer
Journal:  Crit Care Med       Date:  2012-11       Impact factor: 7.598

3.  Polyvariant mutant cystic fibrosis transmembrane conductance regulator genes. The polymorphic (Tg)m locus explains the partial penetrance of the T5 polymorphism as a disease mutation.

Authors:  H Cuppens; W Lin; M Jaspers; B Costes; H Teng; A Vankeerberghen; M Jorissen; G Droogmans; I Reynaert; M Goossens; B Nilius; J J Cassiman
Journal:  J Clin Invest       Date:  1998-01-15       Impact factor: 14.808

4.  Functional characteristics of L1156F-CFTR associated with alcoholic chronic pancreatitis in Japanese.

Authors:  Shiho Kondo; Kotoyo Fujiki; Shigeru B H Ko; Akiko Yamamoto; Miyuki Nakakuki; Yasutomo Ito; Nikolay Shcheynikov; Motoji Kitagawa; Satoru Naruse; Hiroshi Ishiguro
Journal:  Am J Physiol Gastrointest Liver Physiol       Date:  2015-06-18       Impact factor: 4.052

Review 5.  Consensus on the use and interpretation of cystic fibrosis mutation analysis in clinical practice.

Authors:  C Castellani; H Cuppens; M Macek; J J Cassiman; E Kerem; P Durie; E Tullis; B M Assael; C Bombieri; A Brown; T Casals; M Claustres; G R Cutting; E Dequeker; J Dodge; I Doull; P Farrell; C Ferec; E Girodon; M Johannesson; B Kerem; M Knowles; A Munck; P F Pignatti; D Radojkovic; P Rizzotti; M Schwarz; M Stuhrmann; M Tzetis; J Zielenski; J S Elborn
Journal:  J Cyst Fibros       Date:  2008-05       Impact factor: 5.482

6.  Influence of the duplication of CFTR exon 9 and its flanking sequences on diagnosis of cystic fibrosis mutations.

Authors:  Ayman El-Seedy; Tony Dudognon; Frédéric Bilan; Marie-Claude Pasquet; Marie-Pierre Reboul; Albert Iron; Alain Kitzis; Véronique Ladeveze
Journal:  J Mol Diagn       Date:  2009-09       Impact factor: 5.568

7.  Alternative splicing of the ovine CFTR gene.

Authors:  Fiona C Broackes-Carter; Sarah H Williams; Pei Ling Wong; Nathalie Mouchel; Ann Harris
Journal:  Mamm Genome       Date:  2003-11       Impact factor: 2.957

8.  Exon 9 of the CFTR gene: splice site haplotypes and cystic fibrosis mutations.

Authors:  T Dörk; R Fislage; T Neumann; B Wulf; B Tümmler
Journal:  Hum Genet       Date:  1994-01       Impact factor: 4.132

9.  5' exon replacement and repair by spliceosome-mediated RNA trans-splicing.

Authors:  S Gary Mansfield; Rebecca Hawkins Clark; M Puttaraju; Jolanta Kole; Jonathan A Cohn; Lloyd G Mitchell; Mariano A Garcia-Blanco
Journal:  RNA       Date:  2003-10       Impact factor: 4.942

Review 10.  Chronic pancreatitis and cystic fibrosis.

Authors:  H Witt
Journal:  Gut       Date:  2003-05       Impact factor: 23.059

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