Literature DB >> 21322024

The number of CAG repeats within the normal allele does not influence the age of onset in Huntington's disease.

Jiří Klempíř1, Jana Zidovská, Jan Stochl, Věra Kebrdlová Ing, Tereza Uhrová, Jan Roth.   

Abstract

Huntington's disease (HD) is caused by the expansion of the number of CAG repeats on the chromosome 4p16.3, which results in elongated glutamine tract of huntingtin. The purpose of this work was to examine the interaction between the normal and mutant alleles of this gene and their effect on the clinical onset of HD. We hypothesized that in patients with identical number of CAG repeats within the mutant allele, the age of onset of HD is influenced by the number of CAG repeats within the normal allele. We analyzed the relations between the number of CAG repeats within the normal and mutant alleles, the age at HD onset, and the character of initial symptoms in 468 patients with clinically expressed HD. Although the Cox regression coefficient of 0.15 was significant (P < 0.0001), the regression model explained only 28% of the variance of the age at onset related to the effect of the number of CAG repeats within normal allele. Within the groups of patients with the same number of CAG repeats of the mutant allele, number of CAG repeats of the normal allele was found uncorrelated to the age at onset. Furthermore, when analyzing subgroups of patients with the same allelic composition on both alleles, we failed to observe any correlation with the age at the onset. Our analysis gives no corroboration to the idea of a normal allele having a share in the modification of the age at HD onset. We believe that with the current state of knowledge it is not possible to devise a mathematical model for HD onset prediction because too many entirely unknown modifying factors are still involved.
Copyright © 2010 Movement Disorder Society.

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Year:  2010        PMID: 21322024     DOI: 10.1002/mds.23436

Source DB:  PubMed          Journal:  Mov Disord        ISSN: 0885-3185            Impact factor:   10.338


  7 in total

1.  CAG repeat expansion in Huntington disease determines age at onset in a fully dominant fashion.

Authors:  J-M Lee; E M Ramos; J-H Lee; T Gillis; J S Mysore; M R Hayden; S C Warby; P Morrison; M Nance; C A Ross; R L Margolis; F Squitieri; S Orobello; S Di Donato; E Gomez-Tortosa; C Ayuso; O Suchowersky; R J A Trent; E McCusker; A Novelletto; M Frontali; R Jones; T Ashizawa; S Frank; M H Saint-Hilaire; S M Hersch; H D Rosas; D Lucente; M B Harrison; A Zanko; R K Abramson; K Marder; J Sequeiros; J S Paulsen; G B Landwehrmeyer; R H Myers; M E MacDonald; J F Gusella
Journal:  Neurology       Date:  2012-02-08       Impact factor: 9.910

2.  A series of N-terminal epitope tagged Hdh knock-in alleles expressing normal and mutant huntingtin: their application to understanding the effect of increasing the length of normal Huntingtin's polyglutamine stretch on CAG140 mouse model pathogenesis.

Authors:  Shuqiu Zheng; Nima Ghitani; Jessica S Blackburn; Jeh-Ping Liu; Scott O Zeitlin
Journal:  Mol Brain       Date:  2012-08-14       Impact factor: 4.041

3.  Variation within the Huntington's disease gene influences normal brain structure.

Authors:  Mark Mühlau; Juliane Winkelmann; Dan Rujescu; Ina Giegling; Nikolaos Koutsouleris; Christian Gaser; Milan Arsic; Adolph Weindl; Maximilian Reiser; Eva M Meisenzahl
Journal:  PLoS One       Date:  2012-01-03       Impact factor: 3.240

4.  Data Analytics from Enroll-HD, a Global Clinical Research Platform for Huntington's Disease.

Authors:  Georg B Landwehrmeyer; Cheryl J Fitzer-Attas; Joseph D Giuliano; Nilza Gonçalves; Karen E Anderson; Francisco Cardoso; Joaquim J Ferreira; Tiago A Mestre; Julie C Stout; Cristina Sampaio
Journal:  Mov Disord Clin Pract       Date:  2016-06-22

5.  The V471A polymorphism in autophagy-related gene ATG7 modifies age at onset specifically in Italian Huntington disease patients.

Authors:  Silke Metzger; Carolin Walter; Olaf Riess; Raymund A C Roos; Jørgen E Nielsen; David Craufurd; Huu Phuc Nguyen
Journal:  PLoS One       Date:  2013-07-22       Impact factor: 3.240

6.  Examination of Huntington's disease in a Chinese family.

Authors:  Mingxia Yu; Xiaogai Li; Sanyun Wu; Ji Shen; Jiancheng Tu
Journal:  Neural Regen Res       Date:  2014-02-15       Impact factor: 5.135

Review 7.  Neural and mesenchymal stem cells in animal models of Huntington's disease: past experiences and future challenges.

Authors:  Irina Kerkis; Monica Santoro Haddad; Cristiane Wenceslau Valverde; Sabina Glosman
Journal:  Stem Cell Res Ther       Date:  2015-12-14       Impact factor: 6.832

  7 in total

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