Literature DB >> 1349364

Unstable DNA sequence in myotonic dystrophy.

H G Harley1, S A Rundle, W Reardon, J Myring, S Crow, J D Brook, P S Harper, D J Shaw.   

Abstract

A variable DNA sequence has been detected in patients with myotonic dystrophy. We set out to determine whether identification of this specific molecular defect would improve clinical management of patients and families with myotonic dystrophy. 127 affected patients who were studied had an expanded DNA fragment not seen in 73 normal controls. The increase in length of the fragment correlated broadly with disease severity, and we noted expansion of the sequence in successive generations of the same family. Progressive expansion of the affected gene provides a molecular explanation for an apparently earlier onset in successive generations (anticipation) in myotonic dystrophy and supports the role of an unstable repeat sequence as the basis of the defect. The specificity of this finding will assist in accurate diagnosis of myotonic dystrophy and genetic counselling of affected families.

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Year:  1992        PMID: 1349364     DOI: 10.1016/0140-6736(92)90729-m

Source DB:  PubMed          Journal:  Lancet        ISSN: 0140-6736            Impact factor:   79.321


  47 in total

1.  Specific molecular prenatal diagnosis for the CTG mutation in myotonic dystrophy.

Authors:  J Myring; A L Meredith; H G Harley; G Kohn; G Norbury; P S Harper; D J Shaw
Journal:  J Med Genet       Date:  1992-11       Impact factor: 6.318

2.  Instability versus predictability: the molecular diagnosis of myotonic dystrophy.

Authors:  G K Suthers; S M Huson; K E Davies
Journal:  J Med Genet       Date:  1992-11       Impact factor: 6.318

3.  Anatomy of a founder effect: myotonic dystrophy in Northeastern Quebec.

Authors:  Vania Yotova; Damian Labuda; Ewa Zietkiewicz; Dominik Gehl; Alan Lovell; Jean-François Lefebvre; Stéphane Bourgeois; Emilie Lemieux-Blanchard; Marcin Labuda; Hélène Vézina; Louis Houde; Marc Tremblay; Bruno Toupance; Evelyne Heyer; Thomas J Hudson; Claude Laberge
Journal:  Hum Genet       Date:  2005-05-10       Impact factor: 4.132

4.  Allele length of the DMPK CTG repeat is a predictor of progressive myotonic dystrophy type 1 phenotypes.

Authors:  Gayle Overend; Cécilia Légaré; Jean Mathieu; Luigi Bouchard; Cynthia Gagnon; Darren G Monckton
Journal:  Hum Mol Genet       Date:  2019-07-01       Impact factor: 6.150

5.  Identification of a novel protein, DMAP, which interacts with the myotonic dystrophy protein kinase and shows strong homology to D1 snRNP.

Authors:  Y H Fu
Journal:  Genetica       Date:  1996-01       Impact factor: 1.082

Review 6.  Modifiers of CAG/CTG Repeat Instability: Insights from Mammalian Models.

Authors:  Vanessa C Wheeler; Vincent Dion
Journal:  J Huntingtons Dis       Date:  2021

Review 7.  How do C9ORF72 repeat expansions cause amyotrophic lateral sclerosis and frontotemporal dementia: can we learn from other noncoding repeat expansion disorders?

Authors:  Marka van Blitterswijk; Mariely DeJesus-Hernandez; Rosa Rademakers
Journal:  Curr Opin Neurol       Date:  2012-12       Impact factor: 5.710

8.  Myotonic dystrophy: correlation of clinical symptoms with the size of the CTG trinucleotide repeat.

Authors:  A Jaspert; R Fahsold; H Grehl; D Claus
Journal:  J Neurol       Date:  1995-01       Impact factor: 4.849

9.  The natural history of congenital myotonic dystrophy: mortality and long term clinical aspects.

Authors:  W Reardon; R Newcombe; I Fenton; J Sibert; P S Harper
Journal:  Arch Dis Child       Date:  1993-02       Impact factor: 3.791

10.  mtDNA in congenital myotonic dystrophy.

Authors:  D Thyagarajan; E Byrne; A S Noer; P Lertrit; R Kapsa; S Marzuki
Journal:  Am J Hum Genet       Date:  1993-01       Impact factor: 11.025

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