PUBLICATION
Motility phenotype in a zebrafish vmat2 mutant
- Authors
- Sveinsdóttir, H.S., Decker, A., Christensen, C., Lucena, P.B., Þorsteinsson, H., Richert, E., Maier, V.H., Cornell, R., Karlsson, K.Æ.
- ID
- ZDB-PUB-220106-8
- Date
- 2022
- Source
- PLoS One 17: e0259753 (Journal)
- Registered Authors
- Cornell, Robert, Karlsson, Karl, Maier, Valerie Helene, Þorsteinsson, Haraldur
- Keywords
- none
- MeSH Terms
-
- Animals
- Brain/metabolism
- Dopamine/metabolism
- Dopamine Plasma Membrane Transport Proteins/metabolism
- Glial Cell Line-Derived Neurotrophic Factor/metabolism
- Locomotion/genetics*
- Vesicular Monoamine Transport Proteins/genetics*
- Vesicular Monoamine Transport Proteins/metabolism*
- Zebrafish/genetics
- Zebrafish Proteins/genetics
- PubMed
- 34986152 Full text @ PLoS One
Citation
Sveinsdóttir, H.S., Decker, A., Christensen, C., Lucena, P.B., Þorsteinsson, H., Richert, E., Maier, V.H., Cornell, R., Karlsson, K.Æ. (2022) Motility phenotype in a zebrafish vmat2 mutant. PLoS One. 17:e0259753.
Abstract
In the present study, we characterize a novel zebrafish mutant of solute carrier 18A2 (slc18a2), also known as vesicular monoamine transporter 2 (vmat2), that exhibits a behavioural phenotype partially consistent with human Parkinson´s disease. At six days-post-fertilization, behaviour was analysed and demonstrated that vmat2 homozygous mutant larvae, relative to wild types, show changes in motility in a photomotor assay, altered sleep parameters, and reduced dopamine cell number. Following an abrupt lights-off stimulus mutant larvae initiate larger movements but subsequently inhibit them to a lesser extent in comparison to wild-type larvae. Conversely, during a lights-on period, the mutant larvae are hypomotile. Thigmotaxis, a preference to avoid the centre of a behavioural arena, was increased in homozygotes over heterozygotes and wild types, as was daytime sleep ratio. Furthermore, incubating mutant larvae in pramipexole or L-Dopa partially rescued the motor phenotypes, as did injecting glial cell-derived neurotrophic factor (GDNF) into their brains. This novel vmat2 model represents a tool for high throughput pharmaceutical screens for novel therapeutics, in particular those that increase monoamine transport, and for studies of the function of monoamine transporters.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping