Literature DB >> 20640532

Alpha-synuclein loss in spinal muscular atrophy.

Gyula Acsadi1, Xingli Li, Kelley J Murphy, Kathryn J Swoboda, Graham C Parker.   

Abstract

Spinal muscular atrophy, the most prevalent hereditary motor neuron disease, is caused by mutations in the survival motor neuron (SMN) 1 gene. A significant reduction in the encoded SMN protein leads to the degeneration of motor neurons. However, the molecular events leading to this process are not well understood. The present study uses a previously developed neuronal cell culture model of spinal muscular atrophy for a multiplex transcriptome analysis. Furthermore, gene expression analysis was performed on in vitro cell cultures, as well as tissue samples of spinal muscular atrophy patients and transgenic mice. RNA and subsequent Western blot protein analyses suggest that low SMN levels are associated with significantly lower alpha-synuclein expression. Examination of two genes related to vesicular transport showed a similar though less dramatic decrease in expression. The 140-amino acid protein alpha-synuclein, dominant mutations of which have previously been associated with an autosomal dominant form of Parkinson's disease, is strongly expressed in select neurons of the brain. Although not well understood, the physiologic functions of alpha-synuclein have been linked to synaptic vesicular neurotransmitter release and neuroprotection, suggesting a possible contribution to Smn-deficient motor neuron pathology. Furthermore, alpha-synuclein may be a genetic modifier or biomarker of spinal muscular atrophy.

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Year:  2010        PMID: 20640532      PMCID: PMC3918138          DOI: 10.1007/s12031-010-9422-1

Source DB:  PubMed          Journal:  J Mol Neurosci        ISSN: 0895-8696            Impact factor:   3.444


  56 in total

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Authors:  H Braak; U Rüb; D Sandmann-Keil; W P Gai; R A de Vos; E N Jansen Steur; K Arai; E Braak
Journal:  Acta Neuropathol       Date:  2000-05       Impact factor: 17.088

2.  Mice lacking alpha-synuclein display functional deficits in the nigrostriatal dopamine system.

Authors:  A Abeliovich; Y Schmitz; I Fariñas; D Choi-Lundberg; W H Ho; P E Castillo; N Shinsky; J M Verdugo; M Armanini; A Ryan; M Hynes; H Phillips; D Sulzer; A Rosenthal
Journal:  Neuron       Date:  2000-01       Impact factor: 17.173

3.  Synucleins are developmentally expressed, and alpha-synuclein regulates the size of the presynaptic vesicular pool in primary hippocampal neurons.

Authors:  D D Murphy; S M Rueter; J Q Trojanowski; V M Lee
Journal:  J Neurosci       Date:  2000-05-01       Impact factor: 6.167

4.  Expression of alpha-synuclein in non-apoptotic, slowly degenerating facial motoneurones.

Authors:  L B Moran; S Kösel; C Spitzer; F W Schwaiger; O Riess; G W Kreutzberg; M B Graeber
Journal:  J Neurocytol       Date:  2001-06

5.  Stathmin, a microtubule-destabilizing protein, is dysregulated in spinal muscular atrophy.

Authors:  Hsin-Lan Wen; Yuan-Ta Lin; Chen-Hung Ting; Sue Lin-Chao; Hung Li; Hsiu Mei Hsieh-Li
Journal:  Hum Mol Genet       Date:  2010-02-22       Impact factor: 6.150

Review 6.  An update of the mutation spectrum of the survival motor neuron gene (SMN1) in autosomal recessive spinal muscular atrophy (SMA).

Authors:  B Wirth
Journal:  Hum Mutat       Date:  2000       Impact factor: 4.878

Review 7.  Animal models of spinal muscular atrophy.

Authors:  U R Monani; D D Coovert; A H Burghes
Journal:  Hum Mol Genet       Date:  2000-10       Impact factor: 6.150

8.  Cotrafficking of SV2 and synaptotagmin at the synapse.

Authors:  Jia Yao; Amy Nowack; Patricia Kensel-Hammes; Richard G Gardner; Sandra M Bajjalieh
Journal:  J Neurosci       Date:  2010-04-21       Impact factor: 6.167

9.  Valproic acid is neuroprotective in the rotenone rat model of Parkinson's disease: involvement of alpha-synuclein.

Authors:  Barbara Monti; Valentina Gatta; Francesca Piretti; Simonetta S Raffaelli; Marco Virgili; Antonio Contestabile
Journal:  Neurotox Res       Date:  2009-07-21       Impact factor: 3.911

10.  Mitochondrial dysfunction in a neural cell model of spinal muscular atrophy.

Authors:  Gyula Acsadi; Icksoo Lee; Xingli Li; Magomed Khaidakov; Alena Pecinova; Graham C Parker; Maik Hüttemann
Journal:  J Neurosci Res       Date:  2009-09       Impact factor: 4.164

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  7 in total

1.  Protein Mobility Shifts Contribute to Gel Electrophoresis Liquid Chromatography Analysis.

Authors:  Nicholas J Carruthers; Graham C Parker; Theresa Gratsch; Joseph A Caruso; Paul M Stemmer
Journal:  J Biomol Tech       Date:  2015-09

2.  Splicing regulation in spinal muscular atrophy by an RNA structure formed by long-distance interactions.

Authors:  Natalia N Singh; Brian M Lee; Ravindra N Singh
Journal:  Ann N Y Acad Sci       Date:  2015-02-27       Impact factor: 5.691

3.  Proteomic profile of embryonic stem cells with low survival motor neuron protein is consistent with developmental dysfunction.

Authors:  Graham C Parker; Nicholas J Carruthers; Theresa Gratsch; Joseph A Caruso; Paul M Stemmer
Journal:  J Neural Transm (Vienna)       Date:  2016-05-05       Impact factor: 3.575

Review 4.  Invited review: decoding the pathophysiological mechanisms that underlie RNA dysregulation in neurodegenerative disorders: a review of the current state of the art.

Authors:  Matthew J Walsh; Johnathan Cooper-Knock; Jennifer E Dodd; Matthew J Stopford; Simeon R Mihaylov; Janine Kirby; Pamela J Shaw; Guillaume M Hautbergue
Journal:  Neuropathol Appl Neurobiol       Date:  2015-02       Impact factor: 8.090

5.  Comparison of independent screens on differentially vulnerable motor neurons reveals alpha-synuclein as a common modifier in motor neuron diseases.

Authors:  Rachel A Kline; Kevin A Kaifer; Erkan Y Osman; Francesco Carella; Ariana Tiberi; Jolill Ross; Giuseppa Pennetta; Christian L Lorson; Lyndsay M Murray
Journal:  PLoS Genet       Date:  2017-03-31       Impact factor: 5.917

Review 6.  Synucleinopathy in Amyotrophic Lateral Sclerosis: A Potential Avenue for Antisense Therapeutics?

Authors:  Bradley Roberts; Frances Theunissen; Francis L Mastaglia; P Anthony Akkari; Loren L Flynn
Journal:  Int J Mol Sci       Date:  2022-08-19       Impact factor: 6.208

7.  Oxidative Stress Triggers Body-Wide Skipping of Multiple Exons of the Spinal Muscular Atrophy Gene.

Authors:  Joonbae Seo; Natalia N Singh; Eric W Ottesen; Senthilkumar Sivanesan; Maria Shishimorova; Ravindra N Singh
Journal:  PLoS One       Date:  2016-04-25       Impact factor: 3.240

  7 in total

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