2020
DOI: 10.1523/eneuro.0332-19.2020
|View full text |Cite
|
Sign up to set email alerts
|

Early Restoration ofShank3Expression inShank3Knock-Out Mice Prevents Core ASD-Like Behavioral Phenotypes

Abstract: Early restoration of Shank3 expression in Shank3 knockout mice prevents core ASD-like behavioural phenotypes Running Title: Shank3 rescue prevents autism-related phenotypes.

Help me understand this report

Search citation statements

Order By: Relevance

Paper Sections

Select...
2
1
1
1

Citation Types

2
33
0

Year Published

2020
2020
2023
2023

Publication Types

Select...
8
1

Relationship

0
9

Authors

Journals

citations
Cited by 32 publications
(35 citation statements)
references
References 35 publications
2
33
0
Order By: Relevance
“…Nonetheless, the knockdown and rescue approaches have been successful in many instances, including in the field of ASD. Re-expression of Shank3 in a Shank3 conditional knock-in mouse model rescued social interaction deficits and attenuated repetitive grooming behavior in adult mice (Mei et al, 2016); early genetic restoration of WT Shank3 in Shank3 E13 mutant mice rescued the same deficits as well as those in locomotion and rearing (Jaramillo et al, 2020). In MeCP2overexpressing mice, reducing MeCP2 level (e.g., by antisense oligonucleotide treatment) reversed behavioral deficits associated with MECP2 duplication syndrome (Sztainberg et al, 2015).…”
Section: Animal Ndd Models Of Asd With Reduced Pv Expression And/or Dmentioning
confidence: 93%
“…Nonetheless, the knockdown and rescue approaches have been successful in many instances, including in the field of ASD. Re-expression of Shank3 in a Shank3 conditional knock-in mouse model rescued social interaction deficits and attenuated repetitive grooming behavior in adult mice (Mei et al, 2016); early genetic restoration of WT Shank3 in Shank3 E13 mutant mice rescued the same deficits as well as those in locomotion and rearing (Jaramillo et al, 2020). In MeCP2overexpressing mice, reducing MeCP2 level (e.g., by antisense oligonucleotide treatment) reversed behavioral deficits associated with MECP2 duplication syndrome (Sztainberg et al, 2015).…”
Section: Animal Ndd Models Of Asd With Reduced Pv Expression And/or Dmentioning
confidence: 93%
“…reduced to wildtype levels) by restoring Shank3 expression in mice that developed carrying a Shank3 mutation that disrupted most isoforms (Mei et al, 2016). Another recent study showed that an array of behavioral phenotypes could be prevented by rescuing Shank3 expression embryonically (Jaramillo et al, 2020). Taken together with the current study, these results suggest that Shank3 may be required for certain developmental processes, such as synapse formation, that can be triggered later by restoring its expression, but are not lost by disrupting Shank3 expression after development has already occurred.…”
Section: Discussionmentioning
confidence: 99%
“…Cre-expression, and thus deletion of Shank3 in neocortical excitatory neurons (ex4-22|ALL-NEX Cre ), Dlx5/6 -positive GABAergic forebrain neurons (ex4-22|ALL-Dlx5/6 Cre ), which include various subclasses of neocortical interneurons but also MSNs as principal striatal projection neurons [ 169 – 174 ], DRD1- (ex4-22|ALL-Drd1 Cre ) and DRD2-positive neurons (ex4-22|ALL-Drd2 Cre ) [ 129 ], did not induce the phenotype previously observed in the constitutive KO model [ 128 ], though it has to be noted that such deficits of constitutive KO mice were also not replicated in this study. Mixed evidence and even conflicting results concerning social interaction deficits were observed in the models ex9|ANK [ 82 , 175 ], ex13|PDZ [ 139 , 176 ], ex21|PRO [ 125 , 177 179 ], and the rat model ex6|ANK [ 134 , 180 ]. Intact social motivation and interaction was described in the analysis of the models ex4-7|ANK [ 90 ], ex8|ANK-Q321R [ 132 ], ex21|PRO-InsG3728 [ 126 ], and ex11-21|SH3-PRO in rats [ 135 ].…”
Section: Main Textmentioning
confidence: 99%
“…Such deficits of social recognition were observed in the animals of ex4-22|ALL [ 128 ], ex4-7|ANK [ 90 ], ex11|SH3 [ 123 ], ex13|PDZ [ 139 , 176 ], ex21|PRO [ 125 , 178 , 179 ], and the rat models ex6|ANK [ 134 ] or ex11-21|SH3-PRO [ 135 ]. A single study on the model ex21|PRO did not replicate the abovementioned deficits [ 177 ].…”
Section: Main Textmentioning
confidence: 99%