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Article

Disrupted autophagy and neuronal dysfunction in C. elegans knockin models of FUS amyotrophic lateral sclerosis.

Cell reports - 25 Jan 2022

Baskoylu Saba N, Chapkis Natalie, Unsal Burak, Lins Jeremy, Schuch Kelsey, Simon Jonah, Hart Anne C

Abstract excerpt

How mutations in FUS lead to neuronal dysfunction in amyotrophic lateral sclerosis (ALS) patients remains unclear. To examine mechanisms underlying ALS FUS dysfunction, we generate C. elegans knockin models using CRISPR-Cas9-mediated genome editing, creating R524S and P525L ALS FUS models. Although FUS inclusions are not detected, ALS FUS animals show defective neuromuscular function and locomotion under stress....

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Source

Identifiers

PMID: 35081350
DOI: 10.1016/j.celrep.2021.110195

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.

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