Literature DB >> 9463322

Nearby stop codons in exons of the neurofibromatosis type 1 gene are disparate splice effectors.

S Hoffmeyer1, P Nürnberg, H Ritter, R Fahsold, W Leistner, D Kaufmann, W Krone.   

Abstract

Stop mutations are known to disrupt gene function in different ways. They both give rise to truncated polypeptides because of the premature-termination codons (PTCs) and frequently affect the metabolism of the corresponding mRNAs. The analysis of neurofibromin transcripts from different neurofibromatosis type 1 (NF1) patients revealed the skipping of exons containing PTCs. The phenomenon of exon skipping induced by nonsense mutations has been described for other disease genes, including the CFTR (cystic fibrosis transmembrance conductance regulator) gene and the fibrillin gene. We characterized several stop mutations localized within a few base pairs in exons 7 and 37 and noticed complete skipping of either exon in some cases. Because skipping of exon 7 and of exon 37 does not lead to a frameshift, PTCs are avoided in that way. Nuclear-scanning mechanisms for PTCs have been postulated to trigger the removal of the affected exons from the transcript. However, other stop mutations that we found in either NF1 exon did not lead to a skip, although they were localized within the same region. Calculations of minimum-free-energy structures of the respective regions suggest that both changes in the secondary structure of the mRNA and creation or disruption of exonic sequences relevant for the splicing process might in fact cause these different splice phenomena observed in the NF1 gene.

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Year:  1998        PMID: 9463322      PMCID: PMC1376891          DOI: 10.1086/301715

Source DB:  PubMed          Journal:  Am J Hum Genet        ISSN: 0002-9297            Impact factor:   11.025


  46 in total

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Authors:  R Reed
Journal:  Curr Opin Genet Dev       Date:  1996-04       Impact factor: 5.578

2.  Identification of proteins that interact with exon sequences, splice sites, and the branchpoint sequence during each stage of spliceosome assembly.

Authors:  M D Chiara; O Gozani; M Bennett; P Champion-Arnaud; L Palandjian; R Reed
Journal:  Mol Cell Biol       Date:  1996-07       Impact factor: 4.272

3.  A splicing-dependent regulatory mechanism that detects translation signals.

Authors:  M S Carter; S Li; M F Wilkinson
Journal:  EMBO J       Date:  1996-11-01       Impact factor: 11.598

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

5.  New evidence for a mutation hotspot in exon 37 of the NF1 gene.

Authors:  A Böddrich; P N Robinson; M Schülke; A Buske; S Tinschert; P Nürnberg
Journal:  Hum Mutat       Date:  1997       Impact factor: 4.878

6.  A novel mechanism of aberrant pre-mRNA splicing in humans.

Authors:  J D Cogan; M A Prince; S Lekhakula; S Bundey; A Futrakul; E M McCarthy; J A Phillips
Journal:  Hum Mol Genet       Date:  1997-06       Impact factor: 6.150

Review 7.  When cells stop making sense: effects of nonsense codons on RNA metabolism in vertebrate cells.

Authors:  L E Maquat
Journal:  RNA       Date:  1995-07       Impact factor: 4.942

8.  Nonsense mutations inhibit RNA splicing in a cell-free system: recognition of mutant codon is independent of protein synthesis.

Authors:  S Aoufouchi; J Yélamos; C Milstein
Journal:  Cell       Date:  1996-05-03       Impact factor: 41.582

9.  Characterisation of two different nonsense mutations, C6792A and C6792G, causing skipping of exon 37 in the NF1 gene.

Authors:  L Messiaen; T Callens; A De Paepe; M Craen; G Mortier
Journal:  Hum Genet       Date:  1997-11       Impact factor: 4.132

10.  On unequal allelic expression of the neurofibromin gene in neurofibromatosis type 1.

Authors:  S Hoffmeyer; G Assum; J Griesser; D Kaufmann; P Nürnberg; W Krone
Journal:  Hum Mol Genet       Date:  1995-08       Impact factor: 6.150

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  10 in total

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Review 2.  Influence of RNA secondary structure on the pre-mRNA splicing process.

Authors:  Emanuele Buratti; Francisco E Baralle
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Authors:  D Baralle; M Baralle
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4.  Functional analysis of splicing mutations in exon 7 of NF1 gene.

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Journal:  BMC Med Genet       Date:  2007-02-12       Impact factor: 2.103

5.  Minor lesion mutational spectrum of the entire NF1 gene does not explain its high mutability but points to a functional domain upstream of the GAP-related domain.

Authors:  R Fahsold; S Hoffmeyer; C Mischung; C Gille; C Ehlers; N Kücükceylan; M Abdel-Nour; A Gewies; H Peters; D Kaufmann; A Buske; S Tinschert; P Nürnberg
Journal:  Am J Hum Genet       Date:  2000-03       Impact factor: 11.025

6.  NF1 exon 7 skipping and sequence alterations in exonic splice enhancers (ESEs) in a neurofibromatosis 1 patient.

Authors:  Patrizia Colapietro; Cristina Gervasini; Federica Natacci; Livia Rossi; Paola Riva; Lidia Larizza
Journal:  Hum Genet       Date:  2003-09-06       Impact factor: 4.132

7.  Differential expression of splicing variants of the human caldesmon gene (CALD1) in glioma neovascularization versus normal brain microvasculature.

Authors:  Ping-Pin Zheng; Anieta M Sieuwerts; Theo M Luider; M van der Weiden; Peter A E Sillevis-Smitt; Johan M Kros
Journal:  Am J Pathol       Date:  2004-06       Impact factor: 4.307

8.  Splicing defects in the ataxia-telangiectasia gene, ATM: underlying mutations and consequences.

Authors:  S N Teraoka; M Telatar; S Becker-Catania; T Liang; S Onengüt; A Tolun; L Chessa; O Sanal; E Bernatowska; R A Gatti; P Concannon
Journal:  Am J Hum Genet       Date:  1999-06       Impact factor: 11.025

Review 9.  Of numbers and movement - understanding transcription factor pathogenesis by advanced microscopy.

Authors:  Julia M T Auer; Jack J Stoddart; Ioannis Christodoulou; Ana Lima; Kassiani Skouloudaki; Hildegard N Hall; Vladana Vukojević; Dimitrios K Papadopoulos
Journal:  Dis Model Mech       Date:  2020-12-29       Impact factor: 5.758

10.  Functional Analysis of Mutations in Exon 9 of NF1 Reveals the Presence of Several Elements Regulating Splicing.

Authors:  Elisabete Hernández-Imaz; Yolanda Martín; Laura de Conti; German Melean; Ana Valero; Marco Baralle; Concepción Hernández-Chico
Journal:  PLoS One       Date:  2015-10-28       Impact factor: 3.240

  10 in total

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