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Tspyl2 Loss-of-Function Causes Neurodevelopmental Brain and Behavior Abnormalities in Mice
- Source :
- Behavior Genetics, Li, Q, Chan, S Y, Wong, K K, Wei, R, Leung, Y O, Ding, A Y, Hui, T C K, Cheung, C, Chua, S E, Sham, P C, Wu, E X & McAlonan, G M 2016, ' Tspyl2 Loss-of-Function Causes Neurodevelopmental Brain and Behavior Abnormalities in Mice ', Behavior Genetics, pp. 1-9 . https://doi.org/10.1007/s10519-015-9777-8
- Publication Year :
- 2016
- Publisher :
- Springer US, 2016.
-
Abstract
- Testis specific protein, Y-encoded-like 2 (TSPYL2) regulates the expression of genes encoding glutamate receptors. Glutamate pathology is implicated in neurodevelopmental conditions such as autism spectrum disorder, attention deficit hyperactivity disorder (ADHD) and schizophrenia. In line with this, a microduplication incorporating the TSPYL2 locus has been reported in people with ADHD. However, the role of Tspyl2 remains unclear. Therefore here we used a Tspyl2 loss-of-function mouse model to directly examine how this gene impacts upon behavior and brain anatomy. We hypothesized that Tspyl2 knockout (KO) would precipitate a phenotype relevant to neurodevelopmental conditions. In line with this prediction, we found that Tspyl2 KO mice were marginally more active, had significantly impaired prepulse inhibition, and were significantly more ‘sensitive’ to the dopamine agonist amphetamine. In addition, the lateral ventricles were significantly smaller in KO mice. These findings suggest that disrupting Tspyl2 gene expression leads to behavioral and brain morphological alterations that mirror a number of neurodevelopmental psychiatric traits. Electronic supplementary material The online version of this article (doi:10.1007/s10519-015-9777-8) contains supplementary material, which is available to authorized users.
- Subjects :
- 0301 basic medicine
Tspyl2
Male
Cell Cycle Proteins
Motor Activity
Sodium Chloride
Dopamine agonist
Cerebral Ventricles
03 medical and health sciences
Lateral ventricles
0302 clinical medicine
mental disorders
Genetics
medicine
Attention deficit hyperactivity disorder
Animals
Genetics(clinical)
Interpersonal Relations
Amphetamine
Genetics (clinical)
Ecology, Evolution, Behavior and Systematics
Prepulse inhibition
Original Research
Mice, Knockout
Behavior, Animal
Prepulse Inhibition
Glutamate receptor
Brain
Nuclear Proteins
medicine.disease
Phenotype
Magnetic Resonance Imaging
030104 developmental biology
Autism spectrum disorder
Psychology
Neuroscience
030217 neurology & neurosurgery
Locomotion
medicine.drug
MRI
Subjects
Details
- Language :
- English
- ISSN :
- 15733297 and 00018244
- Volume :
- 46
- Database :
- OpenAIRE
- Journal :
- Behavior Genetics
- Accession number :
- edsair.doi.dedup.....77830c5c6bad9bb32aee07162489594b