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Psychiatric-disorder-related behavioral phenotypes and cortical hyperactivity in a mouse model of 3q29 deletion syndrome
https://oist.repo.nii.ac.jp/records/1286
https://oist.repo.nii.ac.jp/records/1286074f8454-277d-4d1e-bf23-74e7a3a4e798
名前 / ファイル | ライセンス | アクション |
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Baba-2019-Psychiatric-disorder-related behavio (3.1 MB)
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Creative Commons Attribution 4.0 International(https://creativecommons.org/licenses/by/4.0/)
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Item type | 学術雑誌論文 / Journal Article(1) | |||||
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公開日 | 2020-03-16 | |||||
タイトル | ||||||
言語 | en | |||||
タイトル | Psychiatric-disorder-related behavioral phenotypes and cortical hyperactivity in a mouse model of 3q29 deletion syndrome | |||||
言語 | ||||||
言語 | eng | |||||
資源タイプ | ||||||
資源タイプ識別子 | http://purl.org/coar/resource_type/c_6501 | |||||
資源タイプ | journal article | |||||
著者(英) |
Baba, Masayuki
× Baba, Masayuki× Yokoyama, Kazumasa× Seiriki, Kaoru× Naka, Yuichiro× Matsumura, Kensuke× Kondo, Momoka× Yamamoto, Kana× Hayashida, Misuzu× Kasai, Atsushi× Ago, Yukio× Nagayasu, Kazuki× Hayata-Takano, Atsuko× Takahashi, Akinori× Yamaguchi, Shun× Mori, Daisuke× Ozaki, Norio× Yamamoto, Tadashi× Takuma, Kazuhiro× Hashimoto, Ryota× Hashimoto, Hitoshi× Nakazawa, Takanobu |
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書誌情報 |
en : Neuropsychopharmacology 巻 44, 号 12, p. 2125-2135, 発行日 2019-06-19 |
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抄録 | ||||||
内容記述タイプ | Other | |||||
内容記述 | 3q29 microdeletion, a rare recurrent copy number variant (CNV), greatly confers an increased risk of psychiatric disorders, such as schizophrenia and autism spectrum disorder (ASD), as well as intellectual disability. However, disease-relevant cellular phenotypes of 3q29 deletion syndrome remain to be identified. To reveal the molecular and cellular etiology of 3q29 deletion syndrome, we generated a mouse model of human 3q29 deletion syndrome by chromosome engineering, which achieved construct validity. 3q29 deletion (Df/+) mice showed reduced body weight and brain volume and, more importantly, impaired social interaction and prepulse inhibition. Importantly, the schizophrenia-related impaired prepulse inhibition was reversed by administration of antipsychotics. These findings are reminiscent of the growth defects and neuropsychiatric behavioral phenotypes in patients with 3q29 deletion syndrome and exemplify that the mouse model achieves some part of face validity and predictive validity. Unbiased whole-brain imaging revealed that neuronal hyperactivation after a behavioral task was strikingly exaggerated in a restricted region of the cortex of Df/+ mice. We further elucidated the cellular phenotypes of neuronal hyperactivation and the reduction of parvalbumin expression in the cortex of Df/+ mice. Thus, the 3q29 mouse model provides invaluable insight into the disease-causative molecular and cellular pathology of psychiatric disorders. | |||||
出版者 | ||||||
出版者 | Springer Nature | |||||
ISSN | ||||||
収録物識別子タイプ | ISSN | |||||
収録物識別子 | 0893-133X | |||||
ISSN | ||||||
収録物識別子タイプ | ISSN | |||||
収録物識別子 | 1740-634X | |||||
PubMed番号 | ||||||
関連タイプ | isIdenticalTo | |||||
識別子タイプ | PMID | |||||
関連識別子 | info:pmid/31216562 | |||||
DOI | ||||||
関連タイプ | isIdenticalTo | |||||
識別子タイプ | DOI | |||||
関連識別子 | info:doi/10.1038/s41386-019-0441-5 | |||||
権利 | ||||||
権利情報 | © 2019 The Author(s). | |||||
情報源 | ||||||
関連名称 | https://creativecommons.org/licenses/by/4.0/ | |||||
関連サイト | ||||||
識別子タイプ | URI | |||||
関連識別子 | https://www.nature.com/articles/s41386-019-0441-5#Abs1 | |||||
著者版フラグ | ||||||
出版タイプ | VoR | |||||
出版タイプResource | http://purl.org/coar/version/c_970fb48d4fbd8a85 |