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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Bibliographic Details
Published in:Experimental Animals 2020, Vol.69(1), pp.26-33
Main Authors: Konishi, Shizuka, Tanaka, Natsuki, Mashimo, Tomoji, Yamamoto, Takashi, Sakuma, Tetsushi, Kaneko, Takehito, Tanaka, Miyuu, Izawa, Takeshi, Yamate, Jyoji, Kuwamura, Mitsuru
Format: Article
Language:English
Subjects:
Ccdc85c
Cerebrum
Gene deletion
Hemorrhage
Hydrocephalus
Mice
Mutation
Nuclease
Original
Phenotypes
rat
Rodents
subcortical heterotopia
Transcription
Transcription activator-like effector nucleases
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Summary: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.
ISSN:1341-1357
1881-7122
DOI:10.1538/expanim.19-0005