Literature DB >> 29462610

Spinal muscular atrophy: Selective motor neuron loss and global defect in the assembly of ribonucleoproteins.

Christine E Beattie1, Stephen J Kolb2.   

Abstract

Spinal muscular atrophy is caused by deletions or mutations in the SMN1 gene that result in reduced expression of the SMN protein. The SMN protein is an essential molecular chaperone that is required for the biogenesis of multiple ribonucleoprotein (RNP) complexes including spliceosomal small nuclear RNPs (snRNPs). Reductions in SMN expression result in a reduced abundance of snRNPs and to downstream RNA splicing alterations. SMN is also present in axons and dendrites and appears to have important roles in the formation of neuronal mRNA-protein complexes during development or neuronal repair. Thus, SMA is an exemplar, selective motor neuron disorder that is caused by defects in fundamental RNA processing events. A detailed molecular understanding of how motor neurons fail, and why other neurons do not, in SMA will yield important principals about motor neuron maintenance and neuronal specificity in neurodegenerative diseases.
Copyright © 2018 Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Local translation; SMN complex; Splicing; Survival motor neuron

Mesh:

Substances:

Year:  2018        PMID: 29462610     DOI: 10.1016/j.brainres.2018.02.022

Source DB:  PubMed          Journal:  Brain Res        ISSN: 0006-8993            Impact factor:   3.252


  3 in total

1.  Motor unit recovery following Smn restoration in mouse models of spinal muscular atrophy.

Authors:  Laura H Comley; Rachel A Kline; Alison K Thomson; Victoria Woschitz; Eric Villalón Landeros; Erkan Y Osman; Christian L Lorson; Lyndsay M Murray
Journal:  Hum Mol Genet       Date:  2022-09-10       Impact factor: 5.121

2.  Intimate functional interactions between TGS1 and the Smn complex revealed by an analysis of the Drosophila eye development.

Authors:  Paolo Maccallini; Francesca Bavasso; Livia Scatolini; Elisabetta Bucciarelli; Gemma Noviello; Veronica Lisi; Valeria Palumbo; Simone D'Angeli; Stefano Cacchione; Giovanni Cenci; Laura Ciapponi; James G Wakefield; Maurizio Gatti; Grazia Daniela Raffa
Journal:  PLoS Genet       Date:  2020-05-26       Impact factor: 5.917

3.  SMA-miRs (miR-181a-5p, -324-5p, and -451a) are overexpressed in spinal muscular atrophy skeletal muscle and serum samples.

Authors:  Emanuela Abiusi; Paola Infante; Lucia Di Marcotullio; Francesco Danilo Tiziano; Cinzia Cagnoli; Ludovica Lospinoso Severini; Marika Pane; Giorgia Coratti; Maria Carmela Pera; Adele D'Amico; Federica Diano; Agnese Novelli; Serena Spartano; Stefania Fiori; Giovanni Baranello; Isabella Moroni; Marina Mora; Maria Barbara Pasanisi; Krizia Pocino; Loredana Le Pera; Davide D'Amico; Lorena Travaglini; Francesco Ria; Claudio Bruno; Denise Locatelli; Enrico Silvio Bertini; Lucia Ovidia Morandi; Eugenio Mercuri
Journal:  Elife       Date:  2021-09-20       Impact factor: 8.140

  3 in total

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