Document Detail


Stiff man-like syndrome and generalized myokymia in spinocerebellar ataxia type 3.
MedLine Citation:
PMID:  16552763     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
We describe the novel association of spinocerebellar ataxia type 3/Machado-Joseph disease (SCA3/MJD) phenotype combining classical clinical presentation and semeiology mimicking stiff man syndrome (SMS). The studied pedigree comprises seven affected members in three generations. Their clinical picture consisted of cerebellar ataxia, pyramidal signs, facial myokymia, and ophthalmoplegia. The proband was a 39-year-old man in whom such a clinical picture, 5 years after onset at age 29, evolved to severe SMS and widespread myokymia. Electrophysiological study revealed continuous muscle activity in proximal limb muscles. Molecular study demonstrated the MJD gene mutation in all four examined patients with 73 to 76 CAG repeats in the expanded allele. We conclude that an excess of motor unit activity including stiff man-like syndrome and widespread myokymia may be an integral part of the SCA3 clinical spectrum.
Authors:
José Berciano; Jon Infante; Antonio García; Carmen de Pablos; Guillermo Amer; José Miguel Polo; Victor Volpini; Onofre Combarros
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Publication Detail:
Type:  Case Reports; Journal Article; Research Support, Non-U.S. Gov't    
Journal Detail:
Title:  Movement disorders : official journal of the Movement Disorder Society     Volume:  21     ISSN:  0885-3185     ISO Abbreviation:  Mov. Disord.     Publication Date:  2006 Jul 
Date Detail:
Created Date:  2006-08-28     Completed Date:  2006-12-15     Revised Date:  -    
Medline Journal Info:
Nlm Unique ID:  8610688     Medline TA:  Mov Disord     Country:  United States    
Other Details:
Languages:  eng     Pagination:  1031-5     Citation Subset:  IM    
Copyright Information:
(c) 2006 Movement Disorder Society.
Affiliation:
Service of Neurology, University Hospital Marqués de Valdecilla, (UC) Santander, Spain. jaberciano@humv.es
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MeSH Terms
Descriptor/Qualifier:
Adult
Aged
Alleles
Atrophy
Brain Stem / pathology
Cerebellum / pathology
Diagnosis, Differential
Disease Progression
Female
Humans
Machado-Joseph Disease / diagnosis,  genetics*
Magnetic Resonance Imaging
Male
Middle Aged
Myokymia / diagnosis,  genetics*
Nerve Tissue Proteins / genetics
Neurologic Examination
Nuclear Proteins / genetics
Ophthalmoplegia / diagnosis,  genetics
Pedigree
Phenotype
Repressor Proteins / genetics
Stiff-Person Syndrome / diagnosis,  genetics*
Tomography, X-Ray Computed
Chemical
Reg. No./Substance:
0/Nerve Tissue Proteins; 0/Nuclear Proteins; 0/Repressor Proteins; EC 3.4.22.-/ATXN3 protein, human

From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine


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