Morphological and Molecular Defects in Human Three-Dimensional Retinal Organoid Model of X-Linked Juvenile Retinoschisis

Kang Chieh Huang, Mong Lien Wang, Shih Jen Chen, Jean Cheng Kuo, Won Jing Wang, Phan Nguyen Nhi Nguyen, Karl J. Wahlin, Jyh Feng Lu, Audrey A. Tran, Michael Shi, Yueh Chien, Aliaksandr A. Yarmishyn, Ping Hsing Tsai, Tien Chun Yang, Wann Neng Jane, Chia Ching Chang, Chi Hsien Peng, Thorsten M. Schlaeger, Shih Hwa Chiou

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52 引文 斯高帕斯(Scopus)

摘要

Chiou, Schlaeger, and colleagues use hiPSC-derived retinal organoids to model X-linked juvenile retinoschisis. They show that patient hiPSC-derived retinal organoids replicate key pathologies observed in patients, including retinal splitting and photoreceptor deficit. The observed abnormalities were normalized in organoids derived from isogenic CRISPR/Cas9 gene-corrected hiPSCs. This validated XLRS in vitro model could be used to test and optimize therapeutic approaches.
原文英語
頁(從 - 到)906-923
頁數18
期刊Stem Cell Reports
13
發行號5
DOIs
出版狀態已發佈 - 11月 12 2019
對外發佈

ASJC Scopus subject areas

  • 生物化學
  • 遺傳學
  • 發展生物學
  • 細胞生物學

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