Kctd13 deletion reduces synaptic transmission via increased RhoA

Escamilla, Christine Ochoa and Filonova, Irina and Walker, Angela K. and Xuan, Zhong X. and Holehonnur, Roopashri and Espinosa, Felipe and Liu, Shunan and Thyme, Summer B. and López-García, Isabel A. and Mendoza, Dorian B. and Usui, Noriyoshi and Ellegood, Jacob and Eisch, Amelia J. and Konopka, Genevieve and Lerch, Jason P. and Schier, Alexander F. and Speed, Haley E. and Powell, Craig M.. (2017) Kctd13 deletion reduces synaptic transmission via increased RhoA. Nature, 551 (7679). pp. 227-231.

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Official URL: https://edoc.unibas.ch/73224/

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Copy-number variants of chromosome 16 region 16p11.2 are linked to neuropsychiatric disorders and are among the most prevalent in autism spectrum disorders. Of many 16p11.2 genes, Kctd13 has been implicated as a major driver of neurodevelopmental phenotypes. The function of KCTD13 in the mammalian brain, however, remains unknown. Here we delete the Kctd13 gene in mice and demonstrate reduced synaptic transmission. Reduced synaptic transmission correlates with increased levels of Ras homolog gene family, member A (RhoA), a KCTD13/CUL3 ubiquitin ligase substrate, and is reversed by RhoA inhibition, suggesting increased RhoA as an important mechanism. In contrast to a previous knockdown study, deletion of Kctd13 or kctd13 does not increase brain size or neurogenesis in mice or zebrafish, respectively. These findings implicate Kctd13 in the regulation of neuronal function relevant to neuropsychiatric disorders and clarify the role of Kctd13 in neurogenesis and brain size. Our data also reveal a potential role for RhoA as a therapeutic target in disorders associated with KCTD13 deletion.
Faculties and Departments:05 Faculty of Science > Departement Biozentrum > Growth & Development > Cell and Developmental Biology (Schier)
UniBasel Contributors:Schier, Alexander
Item Type:Article, refereed
Article Subtype:Research Article
Note:Publication type according to Uni Basel Research Database: Journal article
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Last Modified:31 Mar 2020 13:34
Deposited On:31 Mar 2020 13:34

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