Reduced C9orf72 function leads to defective synaptic vesicle release and neuromuscular dysfunction in zebrafish

Butti et al. generate a C9orf72 loss-of-function model in zebrafish. They find that that C9orf72 is required for presynaptic vesicle trafficking and release at the zebrafish larval neuromuscular junctions. This study provides functional insights into the pathogenesis of amyotrophic lateral sclerosis...

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Main Authors: Zoé Butti, Yingzhou Edward Pan, Jean Giacomotto, Shunmoogum A. Patten
Format: Article
Language:English
Published: Nature Portfolio 2021-06-01
Series:Communications Biology
Online Access:https://doi.org/10.1038/s42003-021-02302-y
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author Zoé Butti
Yingzhou Edward Pan
Jean Giacomotto
Shunmoogum A. Patten
author_facet Zoé Butti
Yingzhou Edward Pan
Jean Giacomotto
Shunmoogum A. Patten
author_sort Zoé Butti
collection DOAJ
description Butti et al. generate a C9orf72 loss-of-function model in zebrafish. They find that that C9orf72 is required for presynaptic vesicle trafficking and release at the zebrafish larval neuromuscular junctions. This study provides functional insights into the pathogenesis of amyotrophic lateral sclerosis and fronto-temporal dementia.
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spelling doaj.art-721401a8b8eb4a4ea4abee48a68082dc2022-12-21T21:27:17ZengNature PortfolioCommunications Biology2399-36422021-06-014111610.1038/s42003-021-02302-yReduced C9orf72 function leads to defective synaptic vesicle release and neuromuscular dysfunction in zebrafishZoé Butti0Yingzhou Edward Pan1Jean Giacomotto2Shunmoogum A. Patten3INRS- Centre Armand-Frappier Santé BiotechnologieINRS- Centre Armand-Frappier Santé BiotechnologieQueensland Brain Institute, University of QueenslandINRS- Centre Armand-Frappier Santé BiotechnologieButti et al. generate a C9orf72 loss-of-function model in zebrafish. They find that that C9orf72 is required for presynaptic vesicle trafficking and release at the zebrafish larval neuromuscular junctions. This study provides functional insights into the pathogenesis of amyotrophic lateral sclerosis and fronto-temporal dementia.https://doi.org/10.1038/s42003-021-02302-y
spellingShingle Zoé Butti
Yingzhou Edward Pan
Jean Giacomotto
Shunmoogum A. Patten
Reduced C9orf72 function leads to defective synaptic vesicle release and neuromuscular dysfunction in zebrafish
Communications Biology
title Reduced C9orf72 function leads to defective synaptic vesicle release and neuromuscular dysfunction in zebrafish
title_full Reduced C9orf72 function leads to defective synaptic vesicle release and neuromuscular dysfunction in zebrafish
title_fullStr Reduced C9orf72 function leads to defective synaptic vesicle release and neuromuscular dysfunction in zebrafish
title_full_unstemmed Reduced C9orf72 function leads to defective synaptic vesicle release and neuromuscular dysfunction in zebrafish
title_short Reduced C9orf72 function leads to defective synaptic vesicle release and neuromuscular dysfunction in zebrafish
title_sort reduced c9orf72 function leads to defective synaptic vesicle release and neuromuscular dysfunction in zebrafish
url https://doi.org/10.1038/s42003-021-02302-y
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AT jeangiacomotto reducedc9orf72functionleadstodefectivesynapticvesiclereleaseandneuromusculardysfunctioninzebrafish
AT shunmoogumapatten reducedc9orf72functionleadstodefectivesynapticvesiclereleaseandneuromusculardysfunctioninzebrafish