Literature DB >> 9270598

Spinobulbar muscular atrophy can mimic ALS: the importance of genetic testing in male patients with atypical ALS.

J S Parboosingh1, D A Figlewicz, A Krizus, V Meininger, N A Azad, D S Newman, G A Rouleau.   

Abstract

The clinical presentation of amyotrophic lateral sclerosis (ALS) is variable and overlaps with that of other motor neuron diseases such as spinobulbar muscular atrophy (SBMA; Kennedy disease). With the identification of disease-specific mutations such as the CAG repeat expansion in the androgen receptor in SBMA, an accurate molecular diagnosis can be made in some patients with motor neuron disease. To determine the extent of misdiagnosis of ALS we screened 147 male ALS patients and 100 unrelated male patients from 100 familial ALS (FALS) kindreds for the presence of the SBMA mutation using polymerase chain reaction methods. We show that ALS was clinically misdiagnosed in 2% of sporadic cases and in two of the 100 FALS kindreds. This study underscores the difficulty in distinguishing SBMA from ALS clinically, particularly in patients who lack the classic signs of each disease.

Entities:  

Mesh:

Year:  1997        PMID: 9270598     DOI: 10.1212/wnl.49.2.568

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


  18 in total

Review 1.  Progress in the pathogenesis of amyotrophic lateral sclerosis.

Authors:  C E Shaw; A al-Chalabi; N Leigh
Journal:  Curr Neurol Neurosci Rep       Date:  2001-01       Impact factor: 5.081

2.  Analysis of inconsistencies in terminology of spinal and bulbar muscular atrophy and its effect on retrieval of research.

Authors:  Shelley Arvin
Journal:  J Med Libr Assoc       Date:  2013-04

Review 3.  Progress in Spinobulbar muscular atrophy research: insights into neuronal dysfunction caused by the polyglutamine-expanded androgen receptor.

Authors:  L K Beitel; T Scanlon; B Gottlieb; M A Trifiro
Journal:  Neurotox Res       Date:  2005       Impact factor: 3.911

4.  Epidemiological survey of X-linked bulbar and spinal muscular atrophy, or Kennedy disease, in the province of Reggio Emilia, Italy.

Authors:  D Guidetti; R Sabadini; A Ferlini; I Torrente
Journal:  Eur J Epidemiol       Date:  2001       Impact factor: 8.082

Review 5.  Pathogenic mechanisms and therapeutic strategies in spinobulbar muscular atrophy.

Authors:  Jason P Chua; Andrew P Lieberman
Journal:  CNS Neurol Disord Drug Targets       Date:  2013-12       Impact factor: 4.388

Review 6.  Spinal and bulbar muscular atrophy: ligand-dependent pathogenesis and therapeutic perspectives.

Authors:  Masahisa Katsuno; Hiroaki Adachi; Fumiaki Tanaka; Gen Sobue
Journal:  J Mol Med (Berl)       Date:  2004-02-27       Impact factor: 4.599

Review 7.  Kennedy's disease (spinal and bulbar muscular atrophy): a clinically oriented review of a rare disease.

Authors:  Marianthi Breza; Georgios Koutsis
Journal:  J Neurol       Date:  2018-07-13       Impact factor: 4.849

8.  Convenient diagnosis of spinal and bulbar muscular atrophy using a microchip electrophoresis system.

Authors:  Hirofumi Maruyama; Hiroyuki Morino; Yuishin Izumi; Kouichi Noda; Hideshi Kawakami
Journal:  Am J Neurodegener Dis       Date:  2013-03-08

9.  Moving toward a predictive and personalized clinical approach in amyotrophic lateral sclerosis: novel developments and future directions in diagnosis, genetics, pathogenesis and therapies.

Authors:  Beatrice Nefussy; Vivian E Drory
Journal:  EPMA J       Date:  2010-06-09       Impact factor: 6.543

10.  Mechanisms mediating spinal and bulbar muscular atrophy: investigations into polyglutamine-expanded androgen receptor function and dysfunction.

Authors:  Lenore K Beitel; Carlos Alvarado; Shaza Mokhtar; Miltiadis Paliouras; Mark Trifiro
Journal:  Front Neurol       Date:  2013-05-15       Impact factor: 4.003

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