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Beyond Motor Neurons in Spinal Muscular Atrophy: A Focus on Neuromuscular Junction.

Authors :
Torri F
Mancuso M
Siciliano G
Ricci G
Source :
International journal of molecular sciences [Int J Mol Sci] 2024 Jul 03; Vol. 25 (13). Date of Electronic Publication: 2024 Jul 03.
Publication Year :
2024

Abstract

5q-Spinal muscular atrophy (5q-SMA) is one of the most common neuromuscular diseases due to homozygous mutations in the SMN1 gene. This leads to a loss of function of the SMN1 gene, which in the end determines lower motor neuron degeneration. Since the generation of the first mouse models of SMA neuropathology, a complex degenerative involvement of the neuromuscular junction and peripheral axons of motor nerves, alongside lower motor neurons, has been described. The involvement of the neuromuscular junction in determining disease symptoms offers a possible parallel therapeutic target. This narrative review aims at providing an overview of the current knowledge about the pathogenesis and significance of neuromuscular junction dysfunction in SMA, circulating biomarkers, outcome measures and available or developing therapeutic approaches.

Details

Language :
English
ISSN :
1422-0067
Volume :
25
Issue :
13
Database :
MEDLINE
Journal :
International journal of molecular sciences
Publication Type :
Academic Journal
Accession number :
39000416
Full Text :
https://doi.org/10.3390/ijms25137311