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Cntnap2 expression in the cerebellum of Foxp2(R552H) mice, with a mutation related to speech-language disorder.
Cntnap2 expression in the cerebellum of Foxp2(R552H) mice, with a mutation related to speech-language disorder.
- Source :
-
Neuroscience letters [Neurosci Lett] 2012 Jan 11; Vol. 506 (2), pp. 277-80. Date of Electronic Publication: 2011 Nov 22. - Publication Year :
- 2012
-
Abstract
- Foxp2(R552H) knock-in (KI) mice carrying a mutation related to human speech-language disorder exhibit impaired ultrasonic vocalization and poor Purkinje cell development. Foxp2 is a forkhead domain-containing transcriptional repressor that associates with its co-repressor CtBP; Foxp2(R552H) displays reduced DNA binding activity. A genetic connection between FOXP2 and CNTNAP2 has been demonstrated in vitro, but not in vivo. Here we show that Cntnap2 mRNA levels significantly increased in the cerebellum of Foxp2(R552H) KI pups, although the cerebellar population of Foxp2-positive Purkinje cells was very small. Furthermore, Cntnap2 immunofluorescence did not decrease in the poorly developed Purkinje cells of Foxp2(R552H) KI pups, although synaptophysin immunofluorescence decreased. Cntnap2 and CtBP were ubiquitously expressed, while Foxp2 co-localized with CtBP only in Purkinje cells. Taken together, these observations suggest that Foxp2 may regulate ultrasonic vocalization by associating with CtBP in Purkinje cells; Cntnap2 may be a target of this co-repressor.<br /> (Copyright © 2011 Elsevier Ireland Ltd. All rights reserved.)
- Subjects :
- Animals
Autistic Disorder complications
Autistic Disorder genetics
Autistic Disorder metabolism
Co-Repressor Proteins
Disease Models, Animal
Fluorescent Antibody Technique
Gene Knock-In Techniques
Language Disorders complications
Language Disorders metabolism
Male
Membrane Proteins genetics
Mice
Mice, Mutant Strains
Nerve Tissue Proteins genetics
Purkinje Cells metabolism
RNA, Messenger analysis
Reverse Transcriptase Polymerase Chain Reaction
Speech Disorders complications
Speech Disorders metabolism
Cerebellum metabolism
Forkhead Transcription Factors genetics
Language Disorders genetics
Membrane Proteins biosynthesis
Mutation
Nerve Tissue Proteins biosynthesis
Repressor Proteins genetics
Speech Disorders genetics
Subjects
Details
- Language :
- English
- ISSN :
- 1872-7972
- Volume :
- 506
- Issue :
- 2
- Database :
- MEDLINE
- Journal :
- Neuroscience letters
- Publication Type :
- Academic Journal
- Accession number :
- 22133810
- Full Text :
- https://doi.org/10.1016/j.neulet.2011.11.022