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SMN, profilin IIa and plastin 3: a link between the deregulation of actin dynamics and SMA pathogenesis.
- Source :
-
Molecular and cellular neurosciences [Mol Cell Neurosci] 2009 Sep; Vol. 42 (1), pp. 66-74. Date of Electronic Publication: 2009 Jun 01. - Publication Year :
- 2009
-
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.
- Subjects :
- Animals
Brain metabolism
Disease Models, Animal
Gene Expression Regulation genetics
Membrane Glycoproteins genetics
Mice
Mice, Inbred C57BL
Mice, Knockout
Microfilament Proteins genetics
Muscular Atrophy, Spinal pathology
Muscular Atrophy, Spinal physiopathology
Profilins genetics
Rats
Spinal Cord metabolism
Survival of Motor Neuron 1 Protein genetics
Transfection methods
Actins metabolism
Gene Expression Regulation physiology
Membrane Glycoproteins metabolism
Microfilament Proteins metabolism
Muscular Atrophy, Spinal metabolism
Profilins metabolism
Survival of Motor Neuron 1 Protein metabolism
Subjects
Details
- Language :
- English
- ISSN :
- 1095-9327
- Volume :
- 42
- Issue :
- 1
- Database :
- MEDLINE
- Journal :
- Molecular and cellular neurosciences
- Publication Type :
- Academic Journal
- Accession number :
- 19497369
- Full Text :
- https://doi.org/10.1016/j.mcn.2009.05.009