Literature DB >> 27460344

Commonality amid diversity: Multi-study proteomic identification of conserved disease mechanisms in spinal muscular atrophy.

Heidi R Fuller1, Thomas H Gillingwater2, Thomas M Wishart3.   

Abstract

The neuromuscular disease spinal muscular atrophy (SMA) is a leading genetic cause of infant mortality, resulting from low levels of full-length survival motor neuron (SMN) protein. Despite having a good understanding of the underlying genetics of SMA, the molecular pathways downstream of SMN that regulate disease pathogenesis remain unclear. The identification of molecular perturbations downstream of SMN is required in order to fully understand the fundamental biological role(s) for SMN in cells and tissues of the body, as well as to develop a range of therapeutic targets for developing novel treatments for SMA. Recent developments in proteomic screening technologies have facilitated proteome-wide investigations of a range of SMA models and tissues, generating novel insights into disease mechanisms by highlighting conserved changes in a range of molecular pathways. Comparative analysis of distinct proteomic datasets reveals conserved changes in pathways converging on GAP43, GAPDH, NCAM, UBA1, LMNA, ANXA2 and COL6A3. Proteomic studies therefore represent a leading tool with which to dissect the molecular mechanisms of disease pathogenesis in SMA, serving to identify potentially attractive targets for the development of novel therapies.
Copyright © 2016 The Authors. Published by Elsevier B.V. All rights reserved.

Entities:  

Keywords:  Proteomics; SMA; SMN; Spinal muscular atrophy; UBA1

Mesh:

Substances:

Year:  2016        PMID: 27460344     DOI: 10.1016/j.nmd.2016.06.004

Source DB:  PubMed          Journal:  Neuromuscul Disord        ISSN: 0960-8966            Impact factor:   4.296


  16 in total

1.  Lamin A/C dysregulation contributes to cardiac pathology in a mouse model of severe spinal muscular atrophy.

Authors:  Darija Šoltić; Hannah K Shorrock; Hazel Allardyce; Emma L Wilson; Ian Holt; Silvia A Synowsky; Sally L Shirran; Simon H Parson; Thomas H Gillingwater; Heidi R Fuller
Journal:  Hum Mol Genet       Date:  2019-11-01       Impact factor: 6.150

2.  A-44G transition in SMN2 intron 6 protects patients with spinal muscular atrophy.

Authors:  Xingxing Wu; Shu-Huei Wang; Junjie Sun; Adrian R Krainer; Yimin Hua; Thomas W Prior
Journal:  Hum Mol Genet       Date:  2017-07-15       Impact factor: 6.150

Review 3.  The phospho-landscape of the survival of motoneuron protein (SMN) protein: relevance for spinal muscular atrophy (SMA).

Authors:  Nora Tula Detering; Tobias Schüning; Niko Hensel; Peter Claus
Journal:  Cell Mol Life Sci       Date:  2022-08-25       Impact factor: 9.207

4.  Developmental and degenerative cardiac defects in the Taiwanese mouse model of severe spinal muscular atrophy.

Authors:  Gillian K Maxwell; Eva Szunyogova; Hannah K Shorrock; Thomas H Gillingwater; Simon H Parson
Journal:  J Anat       Date:  2018-02-22       Impact factor: 2.610

5.  Nusinersen Modulates Proteomics Profiles of Cerebrospinal Fluid in Spinal Muscular Atrophy Type 1 Patients.

Authors:  Laura Bianchi; Maria Sframeli; Lorenza Vantaggiato; Gian Luca Vita; Annamaria Ciranni; Francesca Polito; Rosaria Oteri; Eloisa Gitto; Fabrizio Di Giuseppe; Stefania Angelucci; Antonio Versaci; Sonia Messina; Giuseppe Vita; Luca Bini; M'hammed Aguennouz
Journal:  Int J Mol Sci       Date:  2021-04-21       Impact factor: 5.923

6.  On-chip, multisite extracellular and intracellular recordings from primary cultured skeletal myotubes.

Authors:  Noha Rabieh; Silviya M Ojovan; Nava Shmoel; Hadas Erez; Eilon Maydan; Micha E Spira
Journal:  Sci Rep       Date:  2016-11-04       Impact factor: 4.379

7.  Hyperleptinemia in children with autosomal recessive spinal muscular atrophy type I-III.

Authors:  Heike Kölbel; Berthold P Hauffa; Stefan A Wudy; Anastasios Bouikidis; Adela Della Marina; Ulrike Schara
Journal:  PLoS One       Date:  2017-03-09       Impact factor: 3.240

Review 8.  The role of survival motor neuron protein (SMN) in protein homeostasis.

Authors:  Helena Chaytow; Yu-Ting Huang; Thomas H Gillingwater; Kiterie M E Faller
Journal:  Cell Mol Life Sci       Date:  2018-06-05       Impact factor: 9.261

Review 9.  Molecular Factors Involved in Spinal Muscular Atrophy Pathways as Possible Disease-modifying Candidates.

Authors:  Marianna A Maretina; Galina Y Zheleznyakova; Kristina M Lanko; Anna A Egorova; Vladislav S Baranov; Anton V Kiselev
Journal:  Curr Genomics       Date:  2018-08       Impact factor: 2.236

Review 10.  Mitochondrial Dysfunctions: A Red Thread across Neurodegenerative Diseases.

Authors:  Serena Stanga; Anna Caretto; Marina Boido; Alessandro Vercelli
Journal:  Int J Mol Sci       Date:  2020-05-25       Impact factor: 5.923

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