Cargando…

Modeling neuromuscular diseases in zebrafish

Neuromuscular diseases are a diverse group of conditions that affect the motor system and present some overlapping as well as distinct clinical manifestations. Although individually rare, the combined prevalence of NMDs is similar to Parkinson’s. Over the past decade, new genetic mutations have been...

Descripción completa

Detalles Bibliográficos
Autores principales: Singh, Jaskaran, Patten, Shunmoogum A.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9794147/
https://www.ncbi.nlm.nih.gov/pubmed/36583079
http://dx.doi.org/10.3389/fnmol.2022.1054573
_version_ 1784859977735733248
author Singh, Jaskaran
Patten, Shunmoogum A.
author_facet Singh, Jaskaran
Patten, Shunmoogum A.
author_sort Singh, Jaskaran
collection PubMed
description Neuromuscular diseases are a diverse group of conditions that affect the motor system and present some overlapping as well as distinct clinical manifestations. Although individually rare, the combined prevalence of NMDs is similar to Parkinson’s. Over the past decade, new genetic mutations have been discovered through whole exome/genome sequencing, but the pathogenesis of most NMDs remains largely unexplored. Little information on the molecular mechanism governing the progression and development of NMDs accounts for the continual failure of therapies in clinical trials. Different aspects of the diseases are typically investigated using different models from cells to animals. Zebrafish emerges as an excellent model for studying genetics and pathogenesis and for developing therapeutic interventions for most NMDs. In this review, we describe the generation of different zebrafish genetic models mimicking NMDs and how they are used for drug discovery and therapy development.
format Online
Article
Text
id pubmed-9794147
institution National Center for Biotechnology Information
language English
publishDate 2022
publisher Frontiers Media S.A.
record_format MEDLINE/PubMed
spelling pubmed-97941472022-12-28 Modeling neuromuscular diseases in zebrafish Singh, Jaskaran Patten, Shunmoogum A. Front Mol Neurosci Molecular Neuroscience Neuromuscular diseases are a diverse group of conditions that affect the motor system and present some overlapping as well as distinct clinical manifestations. Although individually rare, the combined prevalence of NMDs is similar to Parkinson’s. Over the past decade, new genetic mutations have been discovered through whole exome/genome sequencing, but the pathogenesis of most NMDs remains largely unexplored. Little information on the molecular mechanism governing the progression and development of NMDs accounts for the continual failure of therapies in clinical trials. Different aspects of the diseases are typically investigated using different models from cells to animals. Zebrafish emerges as an excellent model for studying genetics and pathogenesis and for developing therapeutic interventions for most NMDs. In this review, we describe the generation of different zebrafish genetic models mimicking NMDs and how they are used for drug discovery and therapy development. Frontiers Media S.A. 2022-12-13 /pmc/articles/PMC9794147/ /pubmed/36583079 http://dx.doi.org/10.3389/fnmol.2022.1054573 Text en Copyright © 2022 Singh and Patten. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Molecular Neuroscience
Singh, Jaskaran
Patten, Shunmoogum A.
Modeling neuromuscular diseases in zebrafish
title Modeling neuromuscular diseases in zebrafish
title_full Modeling neuromuscular diseases in zebrafish
title_fullStr Modeling neuromuscular diseases in zebrafish
title_full_unstemmed Modeling neuromuscular diseases in zebrafish
title_short Modeling neuromuscular diseases in zebrafish
title_sort modeling neuromuscular diseases in zebrafish
topic Molecular Neuroscience
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9794147/
https://www.ncbi.nlm.nih.gov/pubmed/36583079
http://dx.doi.org/10.3389/fnmol.2022.1054573
work_keys_str_mv AT singhjaskaran modelingneuromusculardiseasesinzebrafish
AT pattenshunmooguma modelingneuromusculardiseasesinzebrafish