Tspyl2 Loss-of-Function Causes Neurodevelopmental Brain and Behavior Abnormalities in MiceReportar como inadecuado

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Behavior Genetics

, Volume 46, Issue 4, pp 529–537

First Online: 29 January 2016Received: 02 July 2015Accepted: 09 December 2015DOI: 10.1007-s10519-015-9777-8

Cite this article as: Li, Q., Chan, S.Y., Wong, K.K. et al. Behav Genet 2016 46: 529. doi:10.1007-s10519-015-9777-8


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.

KeywordsTspyl2 Prepulse inhibition Locomotion Lateral ventricles MRI Edited by Tamara Phillips.

Electronic supplementary materialThe online version of this article doi:10.1007-s10519-015-9777-8 contains supplementary material, which is available to authorized users.

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Autor: Qi Li - Siu Yuen Chan - Kwun K. Wong - Ran Wei - Yu On Leung - Abby Y. Ding - Tomy C. K. Hui - Charlton Cheung - Sie

Fuente: https://link.springer.com/

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