Literature DB >> 16783570

A novel trinucleotide repeat expansion at chromosome 3q26.2 identified by a CAG/CTG repeat expansion detection array.

S E Holmes1, J S Wentzell, A I Seixas, C Callahan, I Silveira, C A Ross, R L Margolis.   

Abstract

CAG/CTG repeat expansions cause at least 12 different neurological disorders, and additional disorders of this type probably exist. Using the repeat expansion detection (RED) assay, we identified an expanded CAG/CTG repeat in a 50-year-old woman with an autosomal dominant syndrome with prominent progressive sensory neuropathy. The expansion could not be accounted for by any of the CAG/CTG repeats known to undergo expansion. To identify the locus of the expansion, we created a PCR array to assess the repeat length of all repeats of eight or more CAG or CTG triplets in the human genome. The expansion was localized to a repeat contained in an intron of a Genscan-predicted gene, 185 nt downstream of a predicted exon that is conserved through mouse. The closest experimentally verified gene in the region (TNIK, encoding a serine/threonine kinase) occurs approximately 63 Kb downstream from the repeat. The length of the expansion in the proband is 98 triplets. This repeat is not expanded in the proband's cousin (the only other affected family member for whom DNA is currently available) and no expansions were detected in a set of 230 patients with movement disorders of unknown cause. An expanded allele containing 58 triplets was detected in a single control individual, and no other expansions were detected in a set of 255 controls. The normal repeat length ranges from 5 to 30 triplets, with 8 triplets the most common allele. Our results suggest that this new repeat expansion is probably not the direct cause of the phenotype in the proband. Whether the repeat contributes to the patient's phenotype, or is associated with another phenotype, remains to be determined.

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Year:  2006        PMID: 16783570     DOI: 10.1007/s00439-006-0207-0

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


  22 in total

1.  The human genome browser at UCSC.

Authors:  W James Kent; Charles W Sugnet; Terrence S Furey; Krishna M Roskin; Tom H Pringle; Alan M Zahler; David Haussler
Journal:  Genome Res       Date:  2002-06       Impact factor: 9.043

Review 2.  Molecular diagnosis of neurogenetic disorders involving trinucleotide repeat expansions.

Authors:  Ene-Choo Tan; Poh San Lai
Journal:  Expert Rev Mol Diagn       Date:  2005-01       Impact factor: 5.225

3.  FXTAS, SCA10, and SCA17 in American patients with movement disorders.

Authors:  Ana I Seixas; Martin H Maurer; Mark Lin; Colleen Callahan; Alka Ahuja; Tohru Matsuura; Christopher A Ross; Fuki M Hisama; Isabel Silveira; Russell L Margolis
Journal:  Am J Med Genet A       Date:  2005-07-01       Impact factor: 2.802

4.  DNA structure in human RNA polymerase II promoters.

Authors:  A G Pedersen; P Baldi; Y Chauvin; S Brunak
Journal:  J Mol Biol       Date:  1998-08-28       Impact factor: 5.469

5.  Structural basis for triplet repeat disorders: a computational analysis.

Authors:  P Baldi; S Brunak; Y Chauvin; A G Pedersen
Journal:  Bioinformatics       Date:  1999-11       Impact factor: 6.937

6.  Expansion of a novel CAG trinucleotide repeat in the 5' region of PPP2R2B is associated with SCA12.

Authors:  S E Holmes; E E O'Hearn; M G McInnis; D A Gorelick-Feldman; J J Kleiderlein; C Callahan; N G Kwak; R G Ingersoll-Ashworth; M Sherr; A J Sumner; A H Sharp; U Ananth; W K Seltzer; M A Boss; A M Vieria-Saecker; J T Epplen; O Riess; C A Ross; R L Margolis
Journal:  Nat Genet       Date:  1999-12       Impact factor: 38.330

7.  Sequence directed flexibility of DNA and the role of cross-strand hydrogen bonds.

Authors:  D Bhattacharyya; S Kundu; A R Thakur; R Majumdar
Journal:  J Biomol Struct Dyn       Date:  1999-10

8.  Evolutionarily conserved elements in vertebrate, insect, worm, and yeast genomes.

Authors:  Adam Siepel; Gill Bejerano; Jakob S Pedersen; Angie S Hinrichs; Minmei Hou; Kate Rosenbloom; Hiram Clawson; John Spieth; Ladeana W Hillier; Stephen Richards; George M Weinstock; Richard K Wilson; Richard A Gibbs; W James Kent; Webb Miller; David Haussler
Journal:  Genome Res       Date:  2005-07-15       Impact factor: 9.043

9.  Expanded CTG triplet blocks from the myotonic dystrophy gene create the strongest known natural nucleosome positioning elements.

Authors:  Y H Wang; J Griffith
Journal:  Genomics       Date:  1995-01-20       Impact factor: 5.736

10.  Satellog: a database for the identification and prioritization of satellite repeats in disease association studies.

Authors:  Perseus I Missirlis; Carri-Lyn R Mead; Stefanie L Butland; B F Francis Ouellette; Rebecca S Devon; Blair R Leavitt; Robert A Holt
Journal:  BMC Bioinformatics       Date:  2005-06-10       Impact factor: 3.169

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