Literature DB >> 19497369

SMN, profilin IIa and plastin 3: a link between the deregulation of actin dynamics and SMA pathogenesis.

Mélissa Bowerman1, Carrie L Anderson, Ariane Beauvais, Pietro Pilo Boyl, Walter Witke, Rashmi Kothary.   

Abstract

Spinal muscular atrophy (SMA) is the most common human genetic disease resulting in infant mortality. SMA is caused by mutations or deletions in the ubiquitously expressed survival motor neuron 1 (SMN1) gene. Why SMA specifically affects motor neurons remains poorly understood. We have shown that Smn deficient PC12 cells have increased levels of the neuronal profilin IIa protein, leading to an inappropriate activation of the RhoA/ROCK pathway. This suggests that mis-regulation of neuronal actin dynamics is central to SMA pathogenesis. Here, we demonstrate an increase in profilin IIa and a decrease in plastin 3 protein levels in a SMA mouse model. Furthermore, knock-out of profilin II upregulates plastin 3 expression in a Smn-dependent manner. However, the depletion of profilin II and the restoration of plastin 3 are not sufficient to rescue the SMA phenotype. Our study suggests that additional regulators of actin dynamics must also contribute to SMA pathogenesis.

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Year:  2009        PMID: 19497369     DOI: 10.1016/j.mcn.2009.05.009

Source DB:  PubMed          Journal:  Mol Cell Neurosci        ISSN: 1044-7431            Impact factor:   4.314


  47 in total

Review 1.  Spinal muscular atrophy: new and emerging insights from model mice.

Authors:  Gyu-Hwan Park; Shingo Kariya; Umrao R Monani
Journal:  Curr Neurol Neurosci Rep       Date:  2010-03       Impact factor: 5.081

2.  PLS3 expression and SMA phenotype: a commentary on correlation of PLS3 expression with disease severity in children with spinal muscular atrophy.

Authors:  Hisahide Nishio
Journal:  J Hum Genet       Date:  2013-11-28       Impact factor: 3.172

3.  A Drosophila model of spinal muscular atrophy uncouples snRNP biogenesis functions of survival motor neuron from locomotion and viability defects.

Authors:  Kavita Praveen; Ying Wen; A Gregory Matera
Journal:  Cell Rep       Date:  2012-06-21       Impact factor: 9.423

4.  Laminin induced local axonal translation of β-actin mRNA is impaired in SMN-deficient motoneurons.

Authors:  Reena Rathod; Steven Havlicek; Nicolas Frank; Robert Blum; Michael Sendtner
Journal:  Histochem Cell Biol       Date:  2012-07-19       Impact factor: 4.304

5.  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 6.  Diverse role of survival motor neuron protein.

Authors:  Ravindra N Singh; Matthew D Howell; Eric W Ottesen; Natalia N Singh
Journal:  Biochim Biophys Acta Gene Regul Mech       Date:  2017-01-15       Impact factor: 4.490

7.  Bifunctional RNAs targeting the intronic splicing silencer N1 increase SMN levels and reduce disease severity in an animal model of spinal muscular atrophy.

Authors:  Erkan Y Osman; Pei-Fen Yen; Christian L Lorson
Journal:  Mol Ther       Date:  2011-10-25       Impact factor: 11.454

8.  Temporal requirement for SMN in motoneuron development.

Authors:  Le T Hao; Phan Q Duy; James D Jontes; Marc Wolman; Michael Granato; Christine E Beattie
Journal:  Hum Mol Genet       Date:  2013-03-03       Impact factor: 6.150

9.  Neurotransmitter release in motor nerve terminals of a mouse model of mild spinal muscular atrophy.

Authors:  Rocío Ruiz; Lucía Tabares
Journal:  J Anat       Date:  2013-03-13       Impact factor: 2.610

10.  Characterization and in vivo functional analysis of the Schizosaccharomyces pombe ICLN gene.

Authors:  Adrien Barbarossa; Etienne Antoine; Henry Neel; Thierry Gostan; Johann Soret; Rémy Bordonné
Journal:  Mol Cell Biol       Date:  2013-12-02       Impact factor: 4.272

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