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...
Autores principales: | , |
---|---|
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 |