PUBLICATION
fmr1 mutation alters the early development of sensory coding and hunting and social behaviors in larval zebrafish
- Authors
- Zhu, S.I., McCullough, M.H., Pujic, Z., Sibberas, J., Sun, B., Darveniza, T., Bucknall, B., Avitan, L., Goodhill, G.J.
- ID
- ZDB-PUB-230104-15
- Date
- 2023
- Source
- The Journal of neuroscience : the official journal of the Society for Neuroscience 43(7): 1211-1224 (Journal)
- Registered Authors
- Keywords
- none
- MeSH Terms
-
- Animals
- Disease Models, Animal
- Fragile X Mental Retardation Protein/genetics
- Fragile X Syndrome*/genetics
- Hunting
- Larva/metabolism
- Mice, Knockout
- Mutation/genetics
- RNA-Binding Proteins/genetics
- Social Behavior
- Zebrafish*/metabolism
- Zebrafish Proteins/genetics
- Zebrafish Proteins/metabolism
- PubMed
- 36596699 Full text @ J. Neurosci.
Citation
Zhu, S.I., McCullough, M.H., Pujic, Z., Sibberas, J., Sun, B., Darveniza, T., Bucknall, B., Avitan, L., Goodhill, G.J. (2023) fmr1 mutation alters the early development of sensory coding and hunting and social behaviors in larval zebrafish. The Journal of neuroscience : the official journal of the Society for Neuroscience. 43(7):1211-1224.
Abstract
Autism Spectrum Disorders (ASDs) are developmental in origin, however little is known about how they affect the early development of behavior and sensory coding. The most common inherited form of autism is Fragile X syndrome, caused by a mutation in FMR1 Mutation of fmr1 in zebrafish causes anxiety-like behavior, hyperactivity, and hypersensitivity in auditory and visual processing. Here we show that zebrafish fmr1-/- mutant larvae of either sex also display changes in hunting behavior, tectal coding and social interaction. During hunting, they were less successful at catching prey and displayed altered behavioral sequences. In the tectum, representations of prey-like stimuli were more diffuse and had higher dimensionality. In a social behavioral assay they spent more time observing a conspecific, but responded more slowly to social cues. However, when given a choice of rearing environment fmr1-/- larvae preferred one with reduced visual stimulation, and rearing them in this environment reduced genotype-specific effects on tectal excitability. Together these results shed new light on how fmr1-/- changes the early development of neural systems and behavior in a vertebrate.SIGNIFICANCE STATEMENT:Autism spectrum disorders (ASDs) are caused by changes in early neural development. Animal models of ASDs offer the opportunity to study these developmental processes in greater detail than in humans. Here we found that a zebrafish mutant for a gene which in humans causes one type of ASD showed early alterations in hunting behavior, social behavior, and how visual stimuli are represented in the brain. However, we also found that mutant fish preferred reduced visual stimulation, and rearing them in this environment reduced alterations in neural activity patterns. These results suggest interesting new directions for using zebrafish as a model to study the development of brain and behavior in ASDs, and how the impact of ASDs could potentially be reduced.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping