Motor Neuron Abnormalities Correlate with Impaired Movement in Zebrafish that Express Mutant Superoxide Dismutase 1
| Autor: | Caitlin W. Lucas, Kristy C. Yuan, Julie D. Atkin, Madelaine C. Tym, Ian P. Blair, Maxinne Watchon, Hamideh Shahheydari, Alison L. Hogan, Katherine J. Robinson, Garth A. Nicholson, Emily K. Don, Claire Winnick, Angela S. Laird, Nicholas J. Cole |
|---|---|
| Jazyk: | angličtina |
| Rok vydání: | 2019 |
| Předmět: |
chemical screening
endocrine system amyotrophic lateral sclerosis animal structures animal diseases Movement Mutant Danio Behavioral testing Biology Superoxide dismutase Animals Genetically Modified 03 medical and health sciences 0302 clinical medicine Superoxide Dismutase-1 stomatognathic system behavioral testing medicine Animals Amyotrophic lateral sclerosis Zebrafish 030304 developmental biology Motor Neurons 0303 health sciences fungi Original Articles Motor neuron medicine.disease biology.organism_classification Chemical screening Disease Models Animal medicine.anatomical_structure nervous system Mutation biology.protein motor neuron disease Animal Science and Zoology Neuroscience 030217 neurology & neurosurgery Developmental Biology |
| Zdroj: | Zebrafish |
| ISSN: | 1557-8542 1545-8547 |
| Popis: | Amyotrophic lateral sclerosis (ALS) is a fatal neurodegenerative disease characterized by progressive loss of motor neurons. ALS can be modeled in zebrafish (Danio rerio) through the expression of human ALS-causing genes, such as superoxide dismutase 1 (SOD1). Overexpression of mutated human SOD1 protein causes aberrant branching and shortening of spinal motor axons. Despite this, the functional relevance of this axon morphology remains elusive. Our aim was to determine whether this motor axonopathy is correlated with impaired movement in mutant (MT) SOD1-expressing zebrafish. Transgenic zebrafish embryos that express blue fluorescent protein (mTagBFP) in motor neurons were injected with either wild-type (WT) or MT (A4V) human SOD1 messenger ribonucleic acid (mRNA). At 48 hours post-fertilization, larvae movement (distance traveled during behavioral testing) was examined, followed by quantification of motor axon length. Larvae injected with MT SOD1 mRNA had significantly shorter and more aberrantly branched motor axons (p |
| Databáze: | OpenAIRE |
| Externí odkaz: |
|