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  1. 学術雑誌論文
  2. その他
  1. 部局別インデックス
  2. 医学部

Organoids from Nephrotic Disease-Derived iPSCs Identify Impaired NEPHRIN Localization and Slit Diaphragm Formation in Kidney Podocytes

http://hdl.handle.net/20.500.12000/46025
http://hdl.handle.net/20.500.12000/46025
c689aa20-5788-4e77-8f65-95090f44c322
名前 / ファイル ライセンス アクション
1-s2.0-S2213671118303242-main.pdf 1-s2.0-S2213671118303242-main.pdf
Item type デフォルトアイテムタイプ(フル)(1)
公開日 2020-06-04
タイトル
タイトル Organoids from Nephrotic Disease-Derived iPSCs Identify Impaired NEPHRIN Localization and Slit Diaphragm Formation in Kidney Podocytes
言語 en
作成者 Tanigawa, Shunsuke

× Tanigawa, Shunsuke

en Tanigawa, Shunsuke

Islam, Mazharul

× Islam, Mazharul

en Islam, Mazharul

Sharmin, Sazia

× Sharmin, Sazia

en Sharmin, Sazia

Naganuma, Hidekazu

× Naganuma, Hidekazu

en Naganuma, Hidekazu

Yoshimura, Yasuhiro

× Yoshimura, Yasuhiro

en Yoshimura, Yasuhiro

Haque, Fahim

× Haque, Fahim

en Haque, Fahim

Era, Takumi

× Era, Takumi

en Era, Takumi

Nakazato, Hitoshi

× Nakazato, Hitoshi

en Nakazato, Hitoshi

Nakanishi, Koichi

× Nakanishi, Koichi

en Nakanishi, Koichi

Sakuma, Tetsushi

× Sakuma, Tetsushi

en Sakuma, Tetsushi

Yamamoto, Takashi

× Yamamoto, Takashi

en Yamamoto, Takashi

Kurihara, Hidetake

× Kurihara, Hidetake

en Kurihara, Hidetake

Taguchi, Atsuhiro

× Taguchi, Atsuhiro

en Taguchi, Atsuhiro

Nishinakamura, Ryuichi

× Nishinakamura, Ryuichi

en Nishinakamura, Ryuichi

アクセス権
アクセス権 open access
アクセス権URI http://purl.org/coar/access_right/c_abf2
権利情報
言語 en
権利情報 Creative Commons Attribution-NonCommercial-NoDerivatives 4.0
言語 en
権利情報Resource https://creativecommons.org/licenses/by-nc-nd/4.0/
権利情報 https://creativecommons.org/licenses/by-nc-nd/4.0/
内容記述
内容記述タイプ Other
内容記述 Mutations in the NPHS1 gene, which encodes NEPHRIN, cause congenital nephrotic syndrome, resulting from impaired slit diaphragm (SD) formation in glomerular podocytes. However, methods for SD reconstitution have been unavailable, thereby limiting studies in the field. In the present study, we established human induced pluripotent stem cells (iPSCs) from a patient with an NPHS1 missense mutation, and reproduced the SD formation process using iPSC-derived kidney organoids. The mutant NEPHRIN failed to become localized on the cell surface for pre-SD domain formation in the induced podocytes. Upon transplantation, the mutant podocytes developed foot processes, but exhibited impaired SD formation. Genetic correction of the single amino acid mutation restored NEPHRIN localization and phosphorylation, colocalization of other SD-associated proteins, and SD formation. Thus, these kidney organoids from patient-derived iPSCs identified SD abnormalities in the podocytes at the initial phase of congenital nephrotic disease.
内容記述タイプ Other
内容記述 論文
出版者
言語 en
出版者 Cell Press
言語
言語 eng
資源タイプ
資源タイプ journal article
資源タイプ識別子 http://purl.org/coar/resource_type/c_6501
出版タイプ
出版タイプ VoR
出版タイプResource http://purl.org/coar/version/c_970fb48d4fbd8a85
識別子
識別子 http://hdl.handle.net/20.500.12000/46025
識別子タイプ HDL
関連情報
関連識別子
識別子タイプ DOI
関連識別子 https://doi.org/10.1016/j.stemcr.2018.08.003
関連識別子
識別子タイプ DOI
関連識別子 https://doi.org/10.1016/j.stemcr.2018.08.003
収録物識別子
収録物識別子タイプ ISSN
収録物識別子 2213-6711
収録物名
言語 en
収録物名 Stem Cell Reports
書誌情報
巻 11, 号 3, p. 727-740
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