Literature DB >> 8133510

Molecular analysis of late onset Huntington's disease.

B Kremer1, F Squitieri, H Telenius, S E Andrew, J Theilmann, N Spence, Y P Goldberg, M R Hayden.   

Abstract

Late onset Huntington's disease is characterised by onset of symptoms after the age of 50 and is usually associated with a milder course. We have analysed the CAG trinucleotide repeat within the HD gene in 133 late onset patients from 107 extended families. The median upper allele size for the CAG repeat was 42 with a range of 38 to 48 repeats. A significant negative correlation (r = -0.29, p = 0.001) was found between the length of repeat and age of onset for the total cohort. However, for persons with age of onset greater than 60, no significant correlation was found. In addition, no significant correlation was found between age of onset and size of the lower allele and the sex of the affected parent or grandparent. There was no preponderance of maternal descent for late onset cases in this series. This study shows that variation in repeat length only accounts for approximately 7% of the variation in age of onset for persons beyond the age of 50 and clearly shows how with increasing onset age the effect of the repeat length on this onset age seems to diminish.

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Year:  1993        PMID: 8133510      PMCID: PMC1016630          DOI: 10.1136/jmg.30.12.991

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  15 in total

1.  Huntington's chorea in Michigan. I. Demography and genetics.

Authors:  T E REED; J H CHANDLER
Journal:  Am J Hum Genet       Date:  1958-06       Impact factor: 11.025

2.  Analysis of human Y-chromosome-specific reiterated DNA in chromosome variants.

Authors:  L M Kunkel; K D Smith; S H Boyer; D S Borgaonkar; S S Wachtel; O J Miller; W R Breg; H W Jones; J M Rary
Journal:  Proc Natl Acad Sci U S A       Date:  1977-03       Impact factor: 11.205

3.  Some problems in Huntington's chorea.

Authors:  D Cameron; G A Venters
Journal:  Scott Med J       Date:  1967-04       Impact factor: 0.729

4.  Familial correlations for age at onset and age at death in Huntington's disease.

Authors:  C J Brackenridge
Journal:  J Med Genet       Date:  1972-03       Impact factor: 6.318

5.  Huntington's Chorea in South Wales. A genetic and epidemiological study.

Authors:  D A Walker; P S Harper; C E Wells; A Tyler; K Davies; R G Newcombe
Journal:  Clin Genet       Date:  1981-04       Impact factor: 4.438

6.  Parental transmission in Huntington's disease.

Authors:  L N Went; M Vegter-van der Vlis; G W Bruyn
Journal:  Lancet       Date:  1984-05-19       Impact factor: 79.321

7.  Maternal transmission in Huntington's disease.

Authors:  R H Myers; D Goldman; E D Bird; D S Sax; C R Merril; M Schoenfeld; P A Wolf
Journal:  Lancet       Date:  1983-01-29       Impact factor: 79.321

8.  Late-onset variant of Huntington's chorea.

Authors:  E Faught; J C Falgout; D A Leli
Journal:  South Med J       Date:  1983-10       Impact factor: 0.954

9.  A genetic model for age at onset in Huntington disease.

Authors:  L A Farrer; P M Conneally
Journal:  Am J Hum Genet       Date:  1985-03       Impact factor: 11.025

10.  Age-of-onset heterogeneity in Huntington disease families.

Authors:  M A Pericak-Vance; R C Elston; P M Conneally; D V Dawson
Journal:  Am J Med Genet       Date:  1983-01
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  17 in total

Review 1.  Huntington disease--another chapter rewritten.

Authors:  M A Nance
Journal:  Am J Hum Genet       Date:  1996-07       Impact factor: 11.025

2.  A familial factor independent of CAG repeat length influences age at onset of Machado-Joseph disease.

Authors:  A L DeStefano; L A Cupples; P Maciel; C Gaspar; J Radvany; D M Dawson; L Sudarsky; L Corwin; P Coutinho; P MacLeod
Journal:  Am J Hum Genet       Date:  1996-07       Impact factor: 11.025

Review 3.  The genetic defect causing Huntington's disease: repeated in other contexts?

Authors:  J F Gusella; F Persichetti; M E MacDonald
Journal:  Mol Med       Date:  1997-04       Impact factor: 6.354

4.  Tonic mGluR5/CB1-dependent suppression of inhibition as a pathophysiological hallmark in the striatum of mice carrying a mutant form of huntingtin.

Authors:  Anton Dvorzhak; Marcus Semtner; Donald S Faber; Rosemarie Grantyn
Journal:  J Physiol       Date:  2012-12-10       Impact factor: 5.182

5.  Genotypes at the GluR6 kainate receptor locus are associated with variation in the age of onset of Huntington disease.

Authors:  D C Rubinsztein; J Leggo; M Chiano; A Dodge; G Norbury; E Rosser; D Craufurd
Journal:  Proc Natl Acad Sci U S A       Date:  1997-04-15       Impact factor: 11.205

6.  Comparison of mid-age-onset and late-onset Huntington's disease in Finnish patients.

Authors:  Jussi O T Sipilä; Tommi Kauko; Markku Päivärinta; Kari Majamaa
Journal:  J Neurol       Date:  2017-08-28       Impact factor: 4.849

7.  DNA analysis of Huntington's disease in southern Chinese.

Authors:  V Chan; Y L Yu; T P Chan; B Yip; C M Chang; M T Wong; Y W Chan; T K Chan
Journal:  J Med Genet       Date:  1995-02       Impact factor: 6.318

Review 8.  A specific mutation for Huntington's disease.

Authors:  P S Harper
Journal:  J Med Genet       Date:  1993-12       Impact factor: 6.318

9.  Analysis of the 5' upstream sequence of the Huntington's disease (HD) gene shows six new rare alleles which are unrelated to the age at onset of HD.

Authors:  R Coles; J Leggo; D C Rubinsztein
Journal:  J Med Genet       Date:  1997-05       Impact factor: 6.318

10.  Striatal network modeling in Huntington's Disease.

Authors:  Adam Ponzi; Scott J Barton; Kendra D Bunner; Claudia Rangel-Barajas; Emily S Zhang; Benjamin R Miller; George V Rebec; James Kozloski
Journal:  PLoS Comput Biol       Date:  2020-04-17       Impact factor: 4.475

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