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Pathological characteristics of Ccdc85c knockout rats: a rat model of genetic hydrocephalus

Spontaneous hhy mice show hydrocephalus and subcortical heterotopia, and a mutation in the Ccdc85c gene has been identified. To contribute to the comparison of the role of Ccdc85c in different species, we established a Ccdc85c KO rat and investigated its pathological phenotypes. Ccdc85c KO rats were...

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Autores principales: Konishi, Shizuka, Tanaka, Natsuki, Mashimo, Tomoji, Yamamoto, Takashi, Sakuma, Tetsushi, Kaneko, Takehito, Tanaka, Miyuu, Izawa, Takeshi, Yamate, Jyoji, Kuwamura, Mitsuru
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Japanese Association for Laboratory Animal Science 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7004802/
https://www.ncbi.nlm.nih.gov/pubmed/31341137
http://dx.doi.org/10.1538/expanim.19-0005
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author Konishi, Shizuka
Tanaka, Natsuki
Mashimo, Tomoji
Yamamoto, Takashi
Sakuma, Tetsushi
Kaneko, Takehito
Tanaka, Miyuu
Izawa, Takeshi
Yamate, Jyoji
Kuwamura, Mitsuru
author_facet Konishi, Shizuka
Tanaka, Natsuki
Mashimo, Tomoji
Yamamoto, Takashi
Sakuma, Tetsushi
Kaneko, Takehito
Tanaka, Miyuu
Izawa, Takeshi
Yamate, Jyoji
Kuwamura, Mitsuru
author_sort Konishi, Shizuka
collection PubMed
description Spontaneous hhy mice show hydrocephalus and subcortical heterotopia, and a mutation in the Ccdc85c gene has been identified. To contribute to the comparison of the role of Ccdc85c in different species, we established a Ccdc85c KO rat and investigated its pathological phenotypes. Ccdc85c KO rats were produced by genomic engineering using transcription activator-like effector nuclease (TALEN). The KO rats had an approximately 350-bp deletion in Ccdc85c and lacked CCDC85C protein expression. The KO rats showed non-obstructive hydrocephalus, subcortical heterotopia, and intracranial hemorrhage. The KO rats had many pathological characteristics similar to those in hhy mice. These results indicate that CCDC85C plays an important role in cerebral development in rats, and the function of CCDC85C in the cerebrum are similar in rats and mice.
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spelling pubmed-70048022020-02-11 Pathological characteristics of Ccdc85c knockout rats: a rat model of genetic hydrocephalus Konishi, Shizuka Tanaka, Natsuki Mashimo, Tomoji Yamamoto, Takashi Sakuma, Tetsushi Kaneko, Takehito Tanaka, Miyuu Izawa, Takeshi Yamate, Jyoji Kuwamura, Mitsuru Exp Anim Original Spontaneous hhy mice show hydrocephalus and subcortical heterotopia, and a mutation in the Ccdc85c gene has been identified. To contribute to the comparison of the role of Ccdc85c in different species, we established a Ccdc85c KO rat and investigated its pathological phenotypes. Ccdc85c KO rats were produced by genomic engineering using transcription activator-like effector nuclease (TALEN). The KO rats had an approximately 350-bp deletion in Ccdc85c and lacked CCDC85C protein expression. The KO rats showed non-obstructive hydrocephalus, subcortical heterotopia, and intracranial hemorrhage. The KO rats had many pathological characteristics similar to those in hhy mice. These results indicate that CCDC85C plays an important role in cerebral development in rats, and the function of CCDC85C in the cerebrum are similar in rats and mice. Japanese Association for Laboratory Animal Science 2019-07-23 2020 /pmc/articles/PMC7004802/ /pubmed/31341137 http://dx.doi.org/10.1538/expanim.19-0005 Text en ©2020 Japanese Association for Laboratory Animal Science This is an open-access article distributed under the terms of the Creative Commons Attribution Non-Commercial No Derivatives (by-nc-nd) License. (CC-BY-NC-ND 4.0: https://creativecommons.org/licenses/by-nc-nd/4.0/)
spellingShingle Original
Konishi, Shizuka
Tanaka, Natsuki
Mashimo, Tomoji
Yamamoto, Takashi
Sakuma, Tetsushi
Kaneko, Takehito
Tanaka, Miyuu
Izawa, Takeshi
Yamate, Jyoji
Kuwamura, Mitsuru
Pathological characteristics of Ccdc85c knockout rats: a rat model of genetic hydrocephalus
title Pathological characteristics of Ccdc85c knockout rats: a rat model of genetic hydrocephalus
title_full Pathological characteristics of Ccdc85c knockout rats: a rat model of genetic hydrocephalus
title_fullStr Pathological characteristics of Ccdc85c knockout rats: a rat model of genetic hydrocephalus
title_full_unstemmed Pathological characteristics of Ccdc85c knockout rats: a rat model of genetic hydrocephalus
title_short Pathological characteristics of Ccdc85c knockout rats: a rat model of genetic hydrocephalus
title_sort pathological characteristics of ccdc85c knockout rats: a rat model of genetic hydrocephalus
topic Original
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7004802/
https://www.ncbi.nlm.nih.gov/pubmed/31341137
http://dx.doi.org/10.1538/expanim.19-0005
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