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Article

The ALS gene FUS regulates synaptic transmission at the Drosophila neuromuscular junction.

Human molecular genetics - 15 Jul 2014

Machamer James B, Collins Sarah E, Lloyd Thomas E

Abstract excerpt

Mutations in the RNA binding protein Fused in sarcoma (FUS) are estimated to account for 5-10% of all inherited cases of amyotrophic lateral sclerosis (ALS), but the function of FUS in motor neurons is poorly understood. Here, we investigate the early functional consequences of overexpressing wild-type or ALS-associated mutant FUS proteins in Drosophila motor neurons, and compare them to phenotypes arising from...

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Source

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Identifiers

PMID: 24569165
DOI: 10.1093/hmg/ddu094
PMCID: PMC4065154

Genes

FUS

Provenance

PubMed metadata and Literature Corpus relationships. Corpus snapshot updated 7 Sept 2026. Manifest publication-graph-cohort-2026-09-07.1.

Source receipt: pubmed, retrieved 7 Sept 2026.

Related research

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The ALS gene FUS regulates synaptic transmission at the Drosophila neuromuscular junction.PMID 24569165Read ↗

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Defects in Synapse Structure and Function Precede Motor Neuron Degeneration in<i>Drosophila</i>Models of FUS-Related ALSSemantic proximity Read ↗

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Overexpression of an ALS-associated FUS mutation in C. elegans disrupts NMJ morphology and leads to defective neuromuscular transmission.Semantic proximity Read ↗

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Loss and gain of FUS function impair neuromuscular synaptic transmission in a genetic model of ALSSemantic proximity Read ↗

Mitochondrial abnormalities and disruption of the neuromuscular junction precede the clinical phenotype and motor neuron loss in hFUSWT transgenic miceSemantic proximity Read ↗

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ALS-associated mutant FUS induces selective motor neuron degeneration through toxic gain of function.Semantic proximity Read ↗

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