Literature DB >> 14566484

Identification and characterization of a novel splice-site mutation in a patient with Wiskott-Aldrich syndrome.

Nuria Andreu1, Carmen Carreras, Félix Prieto, Xavier Estivill, Victor Volpini, Cristina Fillat.   

Abstract

Wiskott-Aldrich syndrome (WAS) is an X-linked recessive disorder characterized by immunodeficiency, eczema, and thrombocytopenia with small platelets. A wide spectrum of mutations in the Wiskott-Aldrich syndrome protein ( WASP) gene have been identified as causative of the disease. In the present paper, we report on a family with a boy affected by WAS, with a splice-site mutation caused by a T to G substitution in the +2 position of intron 6 (IVS6+2T>G). Expression studies performed in COS-7 and U-937 cells showed that the mutation affected the normal splicing process. As a consequence, an abnormally long transcript of 38 nucleotides is generated. Such missplicing is probably due to the activation of a cryptic splice donor site located 38 nt downstream of exon 6. The translation of such aberrant mRNA will produce a truncated protein with a premature stop at codon 190. Thus, a novel splice-site mutation is reported in a patient with a mild WAS phenotype.

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Year:  2003        PMID: 14566484     DOI: 10.1007/s10038-003-0083-6

Source DB:  PubMed          Journal:  J Hum Genet        ISSN: 1434-5161            Impact factor:   3.172


  14 in total

1.  Identification of WASP mutations in 14 Spanish families with Wiskott-Aldrich syndrome.

Authors:  C Fillat; T Español; M Oset; M Ferrando; X Estivill; V Volpini
Journal:  Am J Med Genet       Date:  2001-04-22

Review 2.  Pre-mRNA splicing and human disease.

Authors:  Nuno André Faustino; Thomas A Cooper
Journal:  Genes Dev       Date:  2003-02-15       Impact factor: 11.361

3.  The mutational spectrum of single base-pair substitutions in mRNA splice junctions of human genes: causes and consequences.

Authors:  M Krawczak; J Reiss; D N Cooper
Journal:  Hum Genet       Date:  1992 Sep-Oct       Impact factor: 4.132

4.  Constitutively activating mutation in WASP causes X-linked severe congenital neutropenia.

Authors:  K Devriendt; A S Kim; G Mathijs; S G Frints; M Schwartz; J J Van Den Oord; G E Verhoef; M A Boogaerts; J P Fryns; D You; M K Rosen; P Vandenberghe
Journal:  Nat Genet       Date:  2001-03       Impact factor: 38.330

Review 5.  The Wiskott-Aldrich syndrome protein: forging the link between actin and cell activation.

Authors:  Karen Badour; Jinyi Zhang; Katherine A Siminovitch
Journal:  Immunol Rev       Date:  2003-04       Impact factor: 12.988

Review 6.  Splicing of messenger RNA precursors.

Authors:  R A Padgett; P J Grabowski; M M Konarska; S Seiler; P A Sharp
Journal:  Annu Rev Biochem       Date:  1986       Impact factor: 23.643

7.  RNA splice junctions of different classes of eukaryotes: sequence statistics and functional implications in gene expression.

Authors:  M B Shapiro; P Senapathy
Journal:  Nucleic Acids Res       Date:  1987-09-11       Impact factor: 16.971

Review 8.  The Wiskott-Aldrich syndrome protein (WASP): roles in signaling and cytoskeletal organization.

Authors:  S B Snapper; F S Rosen
Journal:  Annu Rev Immunol       Date:  1999       Impact factor: 28.527

9.  Construction of a novel database containing aberrant splicing mutations of mammalian genes.

Authors:  K Nakai; H Sakamoto
Journal:  Gene       Date:  1994-04-20       Impact factor: 3.688

10.  The Wiskott-Aldrich syndrome and X-linked congenital thrombocytopenia are caused by mutations of the same gene.

Authors:  Q Zhu; M Zhang; R M Blaese; J M Derry; A Junker; U Francke; S H Chen; H D Ochs
Journal:  Blood       Date:  1995-11-15       Impact factor: 22.113

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  2 in total

1.  A novel Wiskott-Aldrich syndrome protein (WASP) complex mutation identified in a WAS patient results in an aberrant product at the C-terminus from two transcripts with unusual polyA signals.

Authors:  Nuria Andreu; Maricruz García-Rodríguez; Victor Volpini; Cecilia Frecha; Ignacio J Molina; Gumersindo Fontan; Cristina Fillat
Journal:  J Hum Genet       Date:  2005-12-22       Impact factor: 3.172

2.  Platelet actin nodules are podosome-like structures dependent on Wiskott-Aldrich syndrome protein and ARP2/3 complex.

Authors:  Natalie S Poulter; Alice Y Pollitt; Amy Davies; Dessislava Malinova; Gerard B Nash; Mike J Hannon; Zoe Pikramenou; Joshua Z Rappoport; John H Hartwig; Dylan M Owen; Adrian J Thrasher; Stephen P Watson; Steven G Thomas
Journal:  Nat Commun       Date:  2015-06-01       Impact factor: 14.919

  2 in total

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