Literature DB >> 12073025

An Alu-mediated deletion at Xp11.23 leading to Wiskott-Aldrich syndrome.

Maxim I Lutskiy1, Lauren N Jones, Fred S Rosen, Eileen Remold-O'Donnell.   

Abstract

Wiskott-Aldrich syndrome (WAS) is an X-linked disease characterized by thrombocytopenia, eczema and immunodeficiency of varying severity. The WASP gene, mutations of which are responsible for the phenotype, maps to Xp11.23. We describe here a patient with a large deletion in the Xp11.23 region. The deletion, which totals 15.8 kb, begins downstream of DXS1696 and encompasses 13 kb upstream of WASP and includes the distal and proximal promoters and exons 1-6. Analysis of the 5'-boundary region identified sequences missing in the Human Genome database and, as a result, the normal DNA sequence was revised to include 743 bp of novel sequence (AF466616). The patient's upstream breakpoint was localized to an AluSg element within a highly repetitive DNA region containing other Alu elements. A 26-bp recombinogenic element is located downstream of the 5' breakpoint. A 16-bp sequence just upstream of the 5' breakpoint shares close homology with the sequence that spans the 3' breakpoint in intron 6. A heptanucleotide of unknown origin, CAGGGGG, links the 5' and 3' breakpoints. To our knowledge this is the largest deletion in a WAS patient.

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Year:  2002        PMID: 12073025     DOI: 10.1007/s00439-002-0716-4

Source DB:  PubMed          Journal:  Hum Genet        ISSN: 0340-6717            Impact factor:   4.132


  4 in total

1.  Characterization of Alu and recombination-associated motifs mediating a large homozygous SPG7 gene rearrangement causing hereditary spastic paraplegia.

Authors:  Eva López; Carlos Casasnovas; Javier Giménez; Antoni Matilla-Dueñas; Ivelisse Sánchez; Víctor Volpini
Journal:  Neurogenetics       Date:  2014-11-16       Impact factor: 2.660

2.  Reciprocal and nonreciprocal recombination at the glucocerebrosidase gene region: implications for complexity in Gaucher disease.

Authors:  Nahid Tayebi; Barbara K Stubblefield; Joseph K Park; Eduard Orvisky; Jamie M Walker; Mary E LaMarca; Ellen Sidransky
Journal:  Am J Hum Genet       Date:  2003-02-13       Impact factor: 11.025

3.  Heterogeneous duplications in patients with Pelizaeus-Merzbacher disease suggest a mechanism of coupled homologous and nonhomologous recombination.

Authors:  Karen J Woodward; Maria Cundall; Karen Sperle; Erik A Sistermans; Mark Ross; Gareth Howell; Susan M Gribble; Deborah C Burford; Nigel P Carter; Donald L Hobson; James Y Garbern; John Kamholz; Henry Heng; M E Hodes; Sue Malcolm; Grace M Hobson
Journal:  Am J Hum Genet       Date:  2005-10-19       Impact factor: 11.025

4.  Wiskott-Aldrich syndrome protein is required for NK cell cytotoxicity and colocalizes with actin to NK cell-activating immunologic synapses.

Authors:  Jordan S Orange; Narayanaswamy Ramesh; Eileen Remold-O'Donnell; Yoji Sasahara; Louise Koopman; Michael Byrne; Francisco A Bonilla; Fred S Rosen; Raif S Geha; Jack L Strominger
Journal:  Proc Natl Acad Sci U S A       Date:  2002-08-12       Impact factor: 11.205

  4 in total

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