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...
| Main Authors: | , , , , , , , , , , , , , , , , , , , , , , , , |
|---|---|
| Format: | Article |
| Language: | English |
| Published: |
Nature Portfolio
2020-01-01
|
| Series: | Nature Communications |
| Online Access: | https://doi.org/10.1038/s41467-019-14169-z |
| _version_ | 1818991092015562752 |
|---|---|
| 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 |
| collection | DOAJ |
| 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. |
| first_indexed | 2024-12-20T20:04:46Z |
| format | Article |
| id | doaj.art-ed0a4b1f8cee4de29d66e4c76ba8d534 |
| institution | Directory Open Access Journal |
| issn | 2041-1723 |
| language | English |
| last_indexed | 2024-12-20T20:04:46Z |
| publishDate | 2020-01-01 |
| publisher | Nature Portfolio |
| record_format | Article |
| series | Nature Communications |
| spelling | doaj.art-ed0a4b1f8cee4de29d66e4c76ba8d5342022-12-21T19:27:58ZengNature PortfolioNature Communications2041-17232020-01-0111111710.1038/s41467-019-14169-zDstyk mutation leads to congenital scoliosis-like vertebral malformations in zebrafish via dysregulated mTORC1/TFEB pathwayXianding Sun0Yang Zhou1Ruobin Zhang2Zuqiang Wang3Meng Xu4Dali Zhang5Junlan Huang6Fengtao Luo7Fangfang Li8Zhenhong Ni9Siru Zhou10Hangang Chen11Shuai Chen12Liang Chen13Xiaolan Du14Bo Chen15Haiyang Huang16Peng Liu17Liangjun Yin18Juhui Qiu19Di Chen20Chuxia Deng21Yangli Xie22Lingfei Luo23Lin Chen24Department of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityKey Laboratory of Freshwater Fish Reproduction and Development, Ministry of Education, Laboratory of Molecular Developmental Biology, School of Life Sciences, Southwest UniversityDepartment of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityDepartment of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityDepartment of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityDepartment of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityDepartment of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityDepartment of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityDepartment of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityDepartment of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityDepartment of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityDepartment of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityDepartment of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityDepartment of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityDepartment of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityDepartment of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityDepartment of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityDepartment of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityDepartment of Orthopedic Surgery, The Second Affiliated Hospital, Chongqing Medical UniversityCollege of Bioengineering, Chongqing UniversityDepartment of Orthopedic Surgery, Rush University Medical CenterGenetics of Development and Disease Branch, National Institute of Diabetes, Digestive and Kidney Diseases, National Institutes of HealthDepartment of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityKey Laboratory of Freshwater Fish Reproduction and Development, Ministry of Education, Laboratory of Molecular Developmental Biology, School of Life Sciences, Southwest UniversityDepartment of Wound Repair and Rehabilitation, State Key Laboratory of Trauma, Burns and Combined Injury, Trauma Center, Research Institute of Surgery, Daping Hospital, Army Medical UniversityCongenital 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.https://doi.org/10.1038/s41467-019-14169-z |
| spellingShingle | 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 Nature Communications |
| title | 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_short | 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 |
| url | https://doi.org/10.1038/s41467-019-14169-z |
| work_keys_str_mv | AT xiandingsun dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT yangzhou dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT ruobinzhang dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT zuqiangwang dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT mengxu dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT dalizhang dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT junlanhuang dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT fengtaoluo dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT fangfangli dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT zhenhongni dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT siruzhou dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT hangangchen dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT shuaichen dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT liangchen dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT xiaolandu dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT bochen dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT haiyanghuang dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT pengliu dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT liangjunyin dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT juhuiqiu dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT dichen dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT chuxiadeng dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT yanglixie dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT lingfeiluo dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway AT linchen dstykmutationleadstocongenitalscoliosislikevertebralmalformationsinzebrafishviadysregulatedmtorc1tfebpathway |