Dstyk mutation leads to congenital scoliosis-like vertebral malformations in zebrafish via dysregulated mTORC1/TFEB pathway

Congenital scoliosis is a complex genetic disorder characterized by vertebral malformation. Here, the authors demonstrate that loss of dstyk leads to scoliosis in zebrafish due to dysregulated biogenesis of notochord vacuoles and that DSTYK is required for lysosome biogenesis through mTORC1 regulati...

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Autores principales:	Xianding Sun, Yang Zhou, Ruobin Zhang, Zuqiang Wang, Meng Xu, Dali Zhang, Junlan Huang, Fengtao Luo, Fangfang Li, Zhenhong Ni, Siru Zhou, Hangang Chen, Shuai Chen, Liang Chen, Xiaolan Du, Bo Chen, Haiyang Huang, Peng Liu, Liangjun Yin, Juhui Qiu, Di Chen, Chuxia Deng, Yangli Xie, Lingfei Luo, Lin Chen
Formato:	article
Lenguaje:	EN
Publicado:	Nature Portfolio 2020
Materias:	Science Q
Acceso en línea:	https://doaj.org/article/ed0a4b1f8cee4de29d66e4c76ba8d534
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spelling	oai:doaj.org-article:ed0a4b1f8cee4de29d66e4c76ba8d5342021-12-02T15:39:08ZDstyk mutation leads to congenital scoliosis-like vertebral malformations in zebrafish via dysregulated mTORC1/TFEB pathway10.1038/s41467-019-14169-z2041-1723https://doaj.org/article/ed0a4b1f8cee4de29d66e4c76ba8d5342020-01-01T00:00:00Zhttps://doi.org/10.1038/s41467-019-14169-zhttps://doaj.org/toc/2041-1723Congenital scoliosis is a complex genetic disorder characterized by vertebral malformation. Here, the authors demonstrate that loss of dstyk leads to scoliosis in zebrafish due to dysregulated biogenesis of notochord vacuoles and that DSTYK is required for lysosome biogenesis through mTORC1 regulation.Xianding SunYang ZhouRuobin ZhangZuqiang WangMeng XuDali ZhangJunlan HuangFengtao LuoFangfang LiZhenhong NiSiru ZhouHangang ChenShuai ChenLiang ChenXiaolan DuBo ChenHaiyang HuangPeng LiuLiangjun YinJuhui QiuDi ChenChuxia DengYangli XieLingfei LuoLin ChenNature PortfolioarticleScienceQENNature Communications, Vol 11, Iss 1, Pp 1-17 (2020)
institution	DOAJ
collection	DOAJ
language	EN
topic	Science Q
spellingShingle	Science Q Xianding Sun Yang Zhou Ruobin Zhang Zuqiang Wang Meng Xu Dali Zhang Junlan Huang Fengtao Luo Fangfang Li Zhenhong Ni Siru Zhou Hangang Chen Shuai Chen Liang Chen Xiaolan Du Bo Chen Haiyang Huang Peng Liu Liangjun Yin Juhui Qiu Di Chen Chuxia Deng Yangli Xie Lingfei Luo Lin Chen Dstyk mutation leads to congenital scoliosis-like vertebral malformations in zebrafish via dysregulated mTORC1/TFEB pathway
description	Congenital scoliosis is a complex genetic disorder characterized by vertebral malformation. Here, the authors demonstrate that loss of dstyk leads to scoliosis in zebrafish due to dysregulated biogenesis of notochord vacuoles and that DSTYK is required for lysosome biogenesis through mTORC1 regulation.
format	article
author	Xianding Sun Yang Zhou Ruobin Zhang Zuqiang Wang Meng Xu Dali Zhang Junlan Huang Fengtao Luo Fangfang Li Zhenhong Ni Siru Zhou Hangang Chen Shuai Chen Liang Chen Xiaolan Du Bo Chen Haiyang Huang Peng Liu Liangjun Yin Juhui Qiu Di Chen Chuxia Deng Yangli Xie Lingfei Luo Lin Chen
author_facet	Xianding Sun Yang Zhou Ruobin Zhang Zuqiang Wang Meng Xu Dali Zhang Junlan Huang Fengtao Luo Fangfang Li Zhenhong Ni Siru Zhou Hangang Chen Shuai Chen Liang Chen Xiaolan Du Bo Chen Haiyang Huang Peng Liu Liangjun Yin Juhui Qiu Di Chen Chuxia Deng Yangli Xie Lingfei Luo Lin Chen
author_sort	Xianding Sun
title	Dstyk mutation leads to congenital scoliosis-like vertebral malformations in zebrafish via dysregulated mTORC1/TFEB pathway
title_short	Dstyk mutation leads to congenital scoliosis-like vertebral malformations in zebrafish via dysregulated mTORC1/TFEB pathway
title_full	Dstyk mutation leads to congenital scoliosis-like vertebral malformations in zebrafish via dysregulated mTORC1/TFEB pathway
title_fullStr	Dstyk mutation leads to congenital scoliosis-like vertebral malformations in zebrafish via dysregulated mTORC1/TFEB pathway
title_full_unstemmed	Dstyk mutation leads to congenital scoliosis-like vertebral malformations in zebrafish via dysregulated mTORC1/TFEB pathway
title_sort	dstyk mutation leads to congenital scoliosis-like vertebral malformations in zebrafish via dysregulated mtorc1/tfeb pathway
publisher	Nature Portfolio
publishDate	2020
url	https://doaj.org/article/ed0a4b1f8cee4de29d66e4c76ba8d534
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Dstyk mutation leads to congenital scoliosis-like vertebral malformations in zebrafish via dysregulated mTORC1/TFEB pathway

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