Literature DB >> 9536082

Functional analysis of the Huntington's disease (HD) gene promoter.

R Coles1, R Caswell, D C Rubinsztein.   

Abstract

The basis for the highly specific neuronal vulnerability seen in Huntington's disease (HD) has not been determined. Recent studies have demonstrated that variation in HD protein expression occurs in the striatum, with affected regions showing increased HD immunoreactivity. Experiments in HD and SCA1 transgenic mice suggest a correlation between phenotypic severity and expression of the mutant transgene. To gain insights into control of HD gene expression, and to investigate the possibility of cell-cell differences in transcription, we have analysed the 5' upstream region of the HD gene in a neuronal (SK-N-SH) and a non-neuronal (JEG3) cell line. Reporter gene assays demonstrated the presence of a key positive-acting region apparently arising from two Sp1 sites in a tandem repeat acting synergistically. This site is polymorphic, and a single Sp1 site is associated with reduced levels of transcription. These experiments also reveal differences in control of expression between neuronal and non-neuronal cell lines.

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Year:  1998        PMID: 9536082     DOI: 10.1093/hmg/7.5.791

Source DB:  PubMed          Journal:  Hum Mol Genet        ISSN: 0964-6906            Impact factor:   6.150


  13 in total

1.  An upstream open reading frame impedes translation of the huntingtin gene.

Authors:  Joseph Lee; Eun Hee Park; Graeme Couture; Isabelle Harvey; Philippe Garneau; Jerry Pelletier
Journal:  Nucleic Acids Res       Date:  2002-12-01       Impact factor: 16.971

2.  A SNP in the HTT promoter alters NF-κB binding and is a bidirectional genetic modifier of Huntington disease.

Authors:  Kristina Bečanović; Anne Nørremølle; Scott J Neal; Chris Kay; Jennifer A Collins; David Arenillas; Tobias Lilja; Giulia Gaudenzi; Shiana Manoharan; Crystal N Doty; Jessalyn Beck; Nayana Lahiri; Elodie Portales-Casamar; Simon C Warby; Colúm Connolly; Rebecca A G De Souza; Sarah J Tabrizi; Ola Hermanson; Douglas R Langbehn; Michael R Hayden; Wyeth W Wasserman; Blair R Leavitt
Journal:  Nat Neurosci       Date:  2015-05-04       Impact factor: 24.884

3.  A natural antisense transcript at the Huntington's disease repeat locus regulates HTT expression.

Authors:  Daniel W Chung; Dobrila D Rudnicki; Lan Yu; Russell L Margolis
Journal:  Hum Mol Genet       Date:  2011-06-13       Impact factor: 6.150

4.  ESET/SETDB1 gene expression and histone H3 (K9) trimethylation in Huntington's disease.

Authors:  Hoon Ryu; Junghee Lee; Sean W Hagerty; Byoung Yul Soh; Sara E McAlpin; Kerry A Cormier; Karen M Smith; Robert J Ferrante
Journal:  Proc Natl Acad Sci U S A       Date:  2006-12-01       Impact factor: 11.205

Review 5.  Genetics and neuropathology of Huntington's disease.

Authors:  Anton Reiner; Ioannis Dragatsis; Paula Dietrich
Journal:  Int Rev Neurobiol       Date:  2011       Impact factor: 3.230

6.  A molecular investigation of true dominance in Huntington's disease.

Authors:  Y Narain; A Wyttenbach; J Rankin; R A Furlong; D C Rubinsztein
Journal:  J Med Genet       Date:  1999-10       Impact factor: 6.318

7.  Isolation and characterization of the rat huntingtin promoter.

Authors:  C Holzmann; W Mäueler; D Petersohn; T Schmidt; G Thiel; J T Epplen; O Riess
Journal:  Biochem J       Date:  1998-11-15       Impact factor: 3.857

8.  Mouse Huntington's disease homolog mRNA levels: variation and allele effects.

Authors:  Karen T Dixon; Jamie A Cearley; Jesse M Hunter; Peter J Detloff
Journal:  Gene Expr       Date:  2004

9.  Chemotherapy for the brain: the antitumor antibiotic mithramycin prolongs survival in a mouse model of Huntington's disease.

Authors:  Robert J Ferrante; Hoon Ryu; James K Kubilus; Santosh D'Mello; Katharine L Sugars; Junghee Lee; Peiyuan Lu; Karen Smith; Susan Browne; M Flint Beal; Bruce S Kristal; Irina G Stavrovskaya; Sandra Hewett; David C Rubinsztein; Brett Langley; Rajiv R Ratan
Journal:  J Neurosci       Date:  2004-11-17       Impact factor: 6.167

10.  CRISPR/Cas9 Editing of the Mutant Huntingtin Allele In Vitro and In Vivo.

Authors:  Alex Mas Monteys; Shauna A Ebanks; Megan S Keiser; Beverly L Davidson
Journal:  Mol Ther       Date:  2017-01-04       Impact factor: 11.454

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