Cargando…

A long noncoding RNA cluster-based genomic locus maintains proper development and visual function

Long noncoding RNAs (lncRNAs) represent a group of regulatory RNAs that play critical roles in numerous cellular events, but their functional importance in development remains largely unexplored. Here, we discovered a series of previously unidentified gene clusters harboring conserved lncRNAs at the...

Descripción completa

Detalles Bibliográficos
Autores principales: Wang, Fei, Ren, Dalong, Liang, Xiaolin, Ke, Shengwei, Zhang, Bowen, Hu, Bing, Song, Xiaoyuan, Wang, Xiangting
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2019
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6614851/
https://www.ncbi.nlm.nih.gov/pubmed/31127312
http://dx.doi.org/10.1093/nar/gkz444
_version_ 1783433258831183872
author Wang, Fei
Ren, Dalong
Liang, Xiaolin
Ke, Shengwei
Zhang, Bowen
Hu, Bing
Song, Xiaoyuan
Wang, Xiangting
author_facet Wang, Fei
Ren, Dalong
Liang, Xiaolin
Ke, Shengwei
Zhang, Bowen
Hu, Bing
Song, Xiaoyuan
Wang, Xiangting
author_sort Wang, Fei
collection PubMed
description Long noncoding RNAs (lncRNAs) represent a group of regulatory RNAs that play critical roles in numerous cellular events, but their functional importance in development remains largely unexplored. Here, we discovered a series of previously unidentified gene clusters harboring conserved lncRNAs at the nonimprinting regions in brain (CNIBs). Among the seven identified CNIBs, human CNIB1 locus is located at Chr 9q33.3 and conserved from Danio rerio to Homo sapiens. Chr 9q33.3-9q34.11 microdeletion has previously been linked to human nail-patella syndrome (NPS) which is frequently accompanied by developmental and visual deficiencies. By generating CNIB1 deletion alleles in zebrafish, we demonstrated the requirement of CNIB1 for proper growth and development, and visual activities. Furthermore, we found that the role of CNIB1 on visual activity is mediated through a regulator of ocular development-lmx1bb. Collectively, our study shows that CNIB1 lncRNAs are important for zebrafish development and provides an lncRNA cluster-mediated pathophysiological mechanism for human Chr 9q33.3-9q34.11 microdeletion syndrome.
format Online
Article
Text
id pubmed-6614851
institution National Center for Biotechnology Information
language English
publishDate 2019
publisher Oxford University Press
record_format MEDLINE/PubMed
spelling pubmed-66148512019-07-12 A long noncoding RNA cluster-based genomic locus maintains proper development and visual function Wang, Fei Ren, Dalong Liang, Xiaolin Ke, Shengwei Zhang, Bowen Hu, Bing Song, Xiaoyuan Wang, Xiangting Nucleic Acids Res Genomics Long noncoding RNAs (lncRNAs) represent a group of regulatory RNAs that play critical roles in numerous cellular events, but their functional importance in development remains largely unexplored. Here, we discovered a series of previously unidentified gene clusters harboring conserved lncRNAs at the nonimprinting regions in brain (CNIBs). Among the seven identified CNIBs, human CNIB1 locus is located at Chr 9q33.3 and conserved from Danio rerio to Homo sapiens. Chr 9q33.3-9q34.11 microdeletion has previously been linked to human nail-patella syndrome (NPS) which is frequently accompanied by developmental and visual deficiencies. By generating CNIB1 deletion alleles in zebrafish, we demonstrated the requirement of CNIB1 for proper growth and development, and visual activities. Furthermore, we found that the role of CNIB1 on visual activity is mediated through a regulator of ocular development-lmx1bb. Collectively, our study shows that CNIB1 lncRNAs are important for zebrafish development and provides an lncRNA cluster-mediated pathophysiological mechanism for human Chr 9q33.3-9q34.11 microdeletion syndrome. Oxford University Press 2019-07-09 2019-05-25 /pmc/articles/PMC6614851/ /pubmed/31127312 http://dx.doi.org/10.1093/nar/gkz444 Text en © The Author(s) 2019. Published by Oxford University Press on behalf of Nucleic Acids Research. http://creativecommons.org/licenses/by-nc/4.0/ This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0/), which permits non-commercial re-use, distribution, and reproduction in any medium, provided the original work is properly cited. For commercial re-use, please contact journals.permissions@oup.com
spellingShingle Genomics
Wang, Fei
Ren, Dalong
Liang, Xiaolin
Ke, Shengwei
Zhang, Bowen
Hu, Bing
Song, Xiaoyuan
Wang, Xiangting
A long noncoding RNA cluster-based genomic locus maintains proper development and visual function
title A long noncoding RNA cluster-based genomic locus maintains proper development and visual function
title_full A long noncoding RNA cluster-based genomic locus maintains proper development and visual function
title_fullStr A long noncoding RNA cluster-based genomic locus maintains proper development and visual function
title_full_unstemmed A long noncoding RNA cluster-based genomic locus maintains proper development and visual function
title_short A long noncoding RNA cluster-based genomic locus maintains proper development and visual function
title_sort long noncoding rna cluster-based genomic locus maintains proper development and visual function
topic Genomics
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6614851/
https://www.ncbi.nlm.nih.gov/pubmed/31127312
http://dx.doi.org/10.1093/nar/gkz444
work_keys_str_mv AT wangfei alongnoncodingrnaclusterbasedgenomiclocusmaintainsproperdevelopmentandvisualfunction
AT rendalong alongnoncodingrnaclusterbasedgenomiclocusmaintainsproperdevelopmentandvisualfunction
AT liangxiaolin alongnoncodingrnaclusterbasedgenomiclocusmaintainsproperdevelopmentandvisualfunction
AT keshengwei alongnoncodingrnaclusterbasedgenomiclocusmaintainsproperdevelopmentandvisualfunction
AT zhangbowen alongnoncodingrnaclusterbasedgenomiclocusmaintainsproperdevelopmentandvisualfunction
AT hubing alongnoncodingrnaclusterbasedgenomiclocusmaintainsproperdevelopmentandvisualfunction
AT songxiaoyuan alongnoncodingrnaclusterbasedgenomiclocusmaintainsproperdevelopmentandvisualfunction
AT wangxiangting alongnoncodingrnaclusterbasedgenomiclocusmaintainsproperdevelopmentandvisualfunction
AT wangfei longnoncodingrnaclusterbasedgenomiclocusmaintainsproperdevelopmentandvisualfunction
AT rendalong longnoncodingrnaclusterbasedgenomiclocusmaintainsproperdevelopmentandvisualfunction
AT liangxiaolin longnoncodingrnaclusterbasedgenomiclocusmaintainsproperdevelopmentandvisualfunction
AT keshengwei longnoncodingrnaclusterbasedgenomiclocusmaintainsproperdevelopmentandvisualfunction
AT zhangbowen longnoncodingrnaclusterbasedgenomiclocusmaintainsproperdevelopmentandvisualfunction
AT hubing longnoncodingrnaclusterbasedgenomiclocusmaintainsproperdevelopmentandvisualfunction
AT songxiaoyuan longnoncodingrnaclusterbasedgenomiclocusmaintainsproperdevelopmentandvisualfunction
AT wangxiangting longnoncodingrnaclusterbasedgenomiclocusmaintainsproperdevelopmentandvisualfunction