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Aipl1 is required for cone photoreceptor function and survival through the stability of Pde6c and Gc3 in zebrafish
https://oist.repo.nii.ac.jp/records/291
https://oist.repo.nii.ac.jp/records/2918b39787b-6f72-463e-b475-31c17b1e5092
名前 / ファイル | ライセンス | アクション |
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srep45962 (2.9 MB)
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Creative Commons Attribution 4.0 International
(http://creativecommons.org/licenses/by/4.0/) |
Item type | 学術雑誌論文 / Journal Article(1) | |||||
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公開日 | 2018-02-27 | |||||
タイトル | ||||||
言語 | en | |||||
タイトル | Aipl1 is required for cone photoreceptor function and survival through the stability of Pde6c and Gc3 in zebrafish | |||||
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) |
Iribarne, Maria
× Iribarne, Maria× Nishiwaki, Yuko× Nakamura, Shohei× Araragi, Masato× Oguri, Eri× Masai, Ichiro |
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書誌情報 |
en : Scientific Reports 巻 7, p. 45962, 発行日 2017-04-05 |
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抄録 | ||||||
内容記述タイプ | Other | |||||
内容記述 | Genetic mutations in aryl hydrocarbon receptor interacting protein-like 1 (AIPL1) cause photoreceptor degeneration associated with Leber congenital amaurosis 4 (LCA4) in human patients. Here we report retinal phenotypes of a zebrafish aipl1 mutant, gold rush (gosh). In zebrafish, there are two aipl1 genes, aipl1a and aipl1b, which are expressed mainly in rods and cones, respectively. The gosh mutant gene encodes cone-specific aipl1, aipl1b. Cone photoreceptors undergo progressive degeneration in the gosh mutant, indicating that aipl1b is required for cone survival. Furthermore, the cone-specific subunit of cGMP phosphodiesterase 6 (Pde6c) is markedly decreased in the gosh mutant, and the gosh mutation genetically interacts with zebrafish pde6c mutation eclipse (els). These data suggest that Aipl1 is required for Pde6c stability and function. In addition to Pde6c, we found that zebrafish cone-specific guanylate cyclase, zGc3, is also decreased in the gosh and els mutants. Furthermore, zGc3 knockdown embryos showed a marked reduction in Pde6c. These observations illustrate the interdependence of cGMP metabolism regulators between Aipl1, Pde6c, and Gc3 in photoreceptors. |
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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 | 2045-2322 | |||||
PubMed番号 | ||||||
関連タイプ | isIdenticalTo | |||||
識別子タイプ | PMID | |||||
関連識別子 | info:pmid/28378769 | |||||
DOI | ||||||
関連タイプ | isIdenticalTo | |||||
識別子タイプ | DOI | |||||
関連識別子 | info:doi/10.1038/srep45962 | |||||
権利 | ||||||
権利情報 | © 2017 The Author(s) . | |||||
関連サイト | ||||||
識別子タイプ | URI | |||||
関連識別子 | https://www.nature.com/articles/srep45962 | |||||
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 |