Literature DB >> 12130535

A nonsense mutation in the fibrillin-1 gene of a Marfan syndrome patient induces NMD and disrupts an exonic splicing enhancer.

Massimo Caputi1, Raymond J Kendzior, Karen L Beemon.   

Abstract

A nonsense mutation in the fibrillin-1 (FBN1) gene of a Marfan syndrome (MFS) patient induces in-frame exon skipping of FBN1 exon 51. We present evidence, based on both in vivo and in vitro experiments, that the skipping of this exon is due to the disruption of an SC35-dependent splicing enhancer within exon 51. In addition, this nonsense mutation induces nonsense-mediated decay (NMD), which degrades the normally spliced mRNA in the patient's cells. In contrast to NMD, skipping of FBN1 exon 51 does not require translation.

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Year:  2002        PMID: 12130535      PMCID: PMC186389          DOI: 10.1101/gad.997502

Source DB:  PubMed          Journal:  Genes Dev        ISSN: 0890-9369            Impact factor:   11.361


  35 in total

1.  Nonsense codons within the Rous sarcoma virus gag gene decrease the stability of unspliced viral RNA.

Authors:  G F Barker; K Beemon
Journal:  Mol Cell Biol       Date:  1991-05       Impact factor: 4.272

Review 2.  The association of nonsense codons with exon skipping.

Authors:  C R Valentine
Journal:  Mutat Res       Date:  1998-09       Impact factor: 2.433

3.  Nonsense mutations and altered splice-site selection.

Authors:  H C Dietz
Journal:  Am J Hum Genet       Date:  1997-03       Impact factor: 11.025

4.  Silent mutation induces exon skipping of fibrillin-1 gene in Marfan syndrome.

Authors:  W Liu; C Qian; U Francke
Journal:  Nat Genet       Date:  1997-08       Impact factor: 38.330

Review 5.  The regulation of splice-site selection, and its role in human disease.

Authors:  T A Cooper; W Mattox
Journal:  Am J Hum Genet       Date:  1997-08       Impact factor: 11.025

6.  Mutant fibrillin-1 monomers lacking EGF-like domains disrupt microfibril assembly and cause severe marfan syndrome.

Authors:  W Liu; C Qian; K Comeau; T Brenn; H Furthmayr; U Francke
Journal:  Hum Mol Genet       Date:  1996-10       Impact factor: 6.150

7.  Positive control of pre-mRNA splicing in vitro.

Authors:  M Tian; T Maniatis
Journal:  Science       Date:  1992-04-10       Impact factor: 47.728

8.  Disruption of the splicing enhancer sequence within exon 27 of the dystrophin gene by a nonsense mutation induces partial skipping of the exon and is responsible for Becker muscular dystrophy.

Authors:  N Shiga; Y Takeshima; H Sakamoto; K Inoue; Y Yokota; M Yokoyama; M Matsuo
Journal:  J Clin Invest       Date:  1997-11-01       Impact factor: 14.808

9.  Premature chain termination is a unifying mechanism for COL1A1 null alleles in osteogenesis imperfecta type I cell strains.

Authors:  M C Willing; S P Deschenes; R L Slayton; E J Roberts
Journal:  Am J Hum Genet       Date:  1996-10       Impact factor: 11.025

10.  A systematic analysis of the factors that determine the strength of pre-mRNA splicing enhancers.

Authors:  B R Graveley; K J Hertel; T Maniatis
Journal:  EMBO J       Date:  1998-11-16       Impact factor: 11.598

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  35 in total

Review 1.  Nuclear translation: what is the evidence?

Authors:  James E Dahlberg; Elsebet Lund; Elizabeth B Goodwin
Journal:  RNA       Date:  2003-01       Impact factor: 4.942

2.  Stop codon-mediated suppression of splicing is a novel nuclear scanning mechanism not affected by elements of protein synthesis and NMD.

Authors:  Chaim Wachtel; Binghui Li; Joseph Sperling; Ruth Sperling
Journal:  RNA       Date:  2004-09-23       Impact factor: 4.942

3.  Human-specific nonsense mutations identified by genome sequence comparisons.

Authors:  Yoonsoo Hahn; Byungkook Lee
Journal:  Hum Genet       Date:  2006-02-10       Impact factor: 4.132

4.  Alternative splicing induced by nonsense mutations in the immunoglobulin mu VDJ exon is independent of truncation of the open reading frame.

Authors:  Marc Bühler; Oliver Mühlemann
Journal:  RNA       Date:  2004-12-21       Impact factor: 4.942

5.  Nonsense-associated alternative splicing of T-cell receptor beta genes: no evidence for frame dependence.

Authors:  Fabio Mohn; Marc Bühler; Oliver Mühlemann
Journal:  RNA       Date:  2004-12-21       Impact factor: 4.942

6.  Disruption of an exon splicing enhancer in exon 3 of MLH1 is the cause of HNPCC in a Quebec family.

Authors:  S McVety; L Li; P H Gordon; G Chong; W D Foulkes
Journal:  J Med Genet       Date:  2005-05-27       Impact factor: 6.318

7.  Axis inhibition protein 2 (AXIN2) polymorphisms may be a risk factor for selective tooth agenesis.

Authors:  Adrianna Mostowska; Barbara Biedziak; Pawel P Jagodzinski
Journal:  J Hum Genet       Date:  2006-01-24       Impact factor: 3.172

8.  Nonsense codons trigger an RNA partitioning shift.

Authors:  Angela D Bhalla; Jayanthi P Gudikote; Jun Wang; Wai-Kin Chan; Yao-Fu Chang; O Renee Olivas; Miles F Wilkinson
Journal:  J Biol Chem       Date:  2008-12-17       Impact factor: 5.157

9.  Delineation of the mechanisms of aberrant splicing caused by two unusual intronic mutations in the RSK2 gene involved in Coffin-Lowry syndrome.

Authors:  Maria Zeniou; Renata Gattoni; André Hanauer; James Stévenin
Journal:  Nucleic Acids Res       Date:  2004-02-18       Impact factor: 16.971

10.  Frame-disrupting mutations elicit pre-mRNA accumulation independently of frame disruption.

Authors:  J Saadi Imam; Jayanthi P Gudikote; Wai-Kin Chan; Miles F Wilkinson
Journal:  Nucleic Acids Res       Date:  2009-12-09       Impact factor: 16.971

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