Literature DB >> 1461383

Strong correlation between the number of CAG repeats in androgen receptor genes and the clinical onset of features of spinal and bulbar muscular atrophy.

S Igarashi1, Y Tanno, O Onodera, M Yamazaki, S Sato, A Ishikawa, N Miyatani, M Nagashima, Y Ishikawa, K Sahashi.   

Abstract

X-linked spinal and bulbar muscular atrophy (SBMA), a motor neuron disease associated with androgen insensitivity, is caused by androgen receptor gene mutations with an increased number of tandem CAG repeats in exon 1. We investigated the increased number of CAG repeats in androgen receptor genes of 19 SBMA patients and found that this correlated strongly with the age at onset of muscle weakness. Thus, SBMA is the first genetic disease in which a strong correlation between the degree of genetic abnormality (number of CAG tandem repeats) and clinical phenotypic expression is demonstrable. The results further indicate that androgen gene mutation is directly involved in the degeneration of motor neurons.

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Year:  1992        PMID: 1461383     DOI: 10.1212/wnl.42.12.2300

Source DB:  PubMed          Journal:  Neurology        ISSN: 0028-3878            Impact factor:   9.910


  31 in total

Review 1.  Modifiers and mechanisms of multi-system polyglutamine neurodegenerative disorders: lessons from fly models.

Authors:  Moushami Mallik; Subhash C Lakhotia
Journal:  J Genet       Date:  2010-12       Impact factor: 1.166

2.  Molecular analysis of the androgen receptor gene in Hong Kong Chinese infertile men.

Authors:  J Y M Tse; V W S Liu; W S B Yeung; E Y L Lau; E H Y Ng; P C Ho
Journal:  J Assist Reprod Genet       Date:  2003-06       Impact factor: 3.412

Review 3.  Onset Manifestations of Spinal and Bulbar Muscular Atrophy (Kennedy's Disease).

Authors:  Josef Finsterer; Gianni Soraru
Journal:  J Mol Neurosci       Date:  2015-10-19       Impact factor: 3.444

4.  Sensory involvement in X-linked spino-bulbar muscular atrophy (Kennedy's syndrome): an electrophysiological study.

Authors:  A Polo; F Teatini; S D'Anna; P Manganotti; A Salviati; B Dallapiccola; G Zanette; N Rizzuto
Journal:  J Neurol       Date:  1996-05       Impact factor: 4.849

5.  Somatic mosaicism of the expanded CAG trinucleotide repeat in mRNAs for the responsible gene of Machado-Joseph disease (MJD), dentatorubral-pallidoluysian atrophy (DRPLA), and spinal and bulbar muscular atrophy (SBMA).

Authors:  Y Ito; F Tanaka; M Yamamoto; M Doyu; M Nagamatsu; S Riku; T Mitsuma; G Sobue
Journal:  Neurochem Res       Date:  1998-01       Impact factor: 3.996

6.  Expression of androgen receptor in X-linked spinal and bulbar muscular atrophy and amyotrophic lateral sclerosis.

Authors:  A Ogata; T Matsuura; K Tashiro; F Moriwaka; T Demura; T Koyanagi; K Nagashima
Journal:  J Neurol Neurosurg Psychiatry       Date:  1994-10       Impact factor: 10.154

7.  The metabolic and endocrine characteristics in spinal and bulbar muscular atrophy.

Authors:  Angela Rosenbohm; Susanne Hirsch; Alexander E Volk; Torsten Grehl; Julian Grosskreutz; Frank Hanisch; Andreas Herrmann; Katja Kollewe; Wolfram Kress; Thomas Meyer; Susanne Petri; Johannes Prudlo; Carsten Wessig; Hans-Peter Müller; Jens Dreyhaupt; Jochen Weishaupt; Christian Kubisch; Jan Kassubek; Patrick Weydt; Albert C Ludolph
Journal:  J Neurol       Date:  2018-02-20       Impact factor: 4.849

8.  Kennedy's disease: clinical and molecular study of two Italian families.

Authors:  D Pareyson; B Castellotti; S Botti; C A Defanti; C Gellera; F Taroni; A Sghirlanzoni
Journal:  Ital J Neurol Sci       Date:  1995-10

9.  CHIP overexpression reduces mutant androgen receptor protein and ameliorates phenotypes of the spinal and bulbar muscular atrophy transgenic mouse model.

Authors:  Hiroaki Adachi; Masahiro Waza; Keisuke Tokui; Masahisa Katsuno; Makoto Minamiyama; Fumiaki Tanaka; Manabu Doyu; Gen Sobue
Journal:  J Neurosci       Date:  2007-05-09       Impact factor: 6.167

10.  Decrease in androgen binding and effect of androgen treatment in a case of X-linked bulbospinal neuronopathy.

Authors:  A Danek; T N Witt; K Mann; H U Schweikert; G Romalo; A R La Spada; K H Fischbeck
Journal:  Clin Investig       Date:  1994-11
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