Document Detail


Synphilin-1 attenuates neuronal degeneration in the A53T alpha-synuclein transgenic mouse model.
MedLine Citation:
PMID:  20185556     Owner:  NLM     Status:  MEDLINE    
Abstract/OtherAbstract:
Genetic alterations in alpha-synuclein cause autosomal dominant familial Parkinsonism and may contribute to sporadic Parkinson's disease (PD). Synphilin-1 is an alpha-synuclein-interacting protein, with implications in PD pathogenesis related to protein aggregation. Currently, the in vivo role of synphilin-1 in alpha-synuclein-linked pathogenesis is not fully understood. Using the mouse prion protein promoter, we generated synphilin-1 transgenic mice, which did not display PD-like phenotypes. However, synphilin-1/A53T alpha-synuclein double-transgenic mice survived longer than A53T alpha-synuclein single-transgenic mice. There were attenuated A53T alpha-synuclein-induced motor abnormalities and decreased astroglial reaction and neuronal degeneration in brains in double-transgenic mice. Overexpression of synphilin-1 decreased caspase-3 activation, increased beclin-1 and LC3 II expression and promoted formation of aggresome-like structures, suggesting that synphilin-1 alters multiple cellular pathways to protect against neuronal degeneration. These studies demonstrate that synphilin-1 can diminish the severity of alpha-synucleinopathy and play a neuroprotective role against A53T alpha-synuclein toxicity in vivo.
Authors:
Wanli W Smith; Zhaohui Liu; Yideng Liang; Naoki Masuda; Debbie A Swing; Nancy A Jenkins; Neal G Copeland; Juan C Troncoso; Mikhail Pletnikov; Ted M Dawson; Lee J Martin; Timothy H Moran; Michael K Lee; David R Borchelt; Christopher A Ross
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Publication Detail:
Type:  Journal Article; Research Support, N.I.H., Extramural     Date:  2010-02-25
Journal Detail:
Title:  Human molecular genetics     Volume:  19     ISSN:  1460-2083     ISO Abbreviation:  Hum. Mol. Genet.     Publication Date:  2010 Jun 
Date Detail:
Created Date:  2010-05-07     Completed Date:  2010-09-22     Revised Date:  2011-07-28    
Medline Journal Info:
Nlm Unique ID:  9208958     Medline TA:  Hum Mol Genet     Country:  England    
Other Details:
Languages:  eng     Pagination:  2087-98     Citation Subset:  IM    
Affiliation:
Division of Neurobiology, Johns Hopkins University School of Medicine, Baltimore, MD 21287, USA.
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MeSH Terms
Descriptor/Qualifier:
Analysis of Variance
Animals
Apoptosis Regulatory Proteins / metabolism
Brain / metabolism,  pathology*
Carrier Proteins / genetics*,  metabolism
Caspase 3 / metabolism
Immunoblotting
Immunohistochemistry
Lewy Bodies / metabolism
Mice
Mice, Transgenic
Mutation, Missense / genetics
Nerve Degeneration / etiology,  metabolism*
Nerve Tissue Proteins / genetics*,  metabolism
Parkinson Disease / complications,  genetics*
alpha-Synuclein / genetics*
Grant Support
ID/Acronym/Agency:
NS38377/NS/NINDS NIH HHS; R01NS055252/NS/NINDS NIH HHS
Chemical
Reg. No./Substance:
0/Apoptosis Regulatory Proteins; 0/Becn1 protein, mouse; 0/Carrier Proteins; 0/Nerve Tissue Proteins; 0/Sncaip protein, mouse; 0/alpha-Synuclein; EC 3.4.22.-/Casp3 protein, mouse; EC 3.4.22.-/Caspase 3
Comments/Corrections

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


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