PUBLICATION

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

Authors
Wang, F., Ren, D., Liang, X., Ke, S., Zhang, B., Hu, B., Song, X., Wang, X.
ID
ZDB-PUB-190528-12
Date
2019
Source
Nucleic acids research   47(12): 6315-6329 (Journal)
Registered Authors
Hu, Bing
Keywords
none
MeSH Terms
  • Male
  • Chromosome Deletion
  • RNA, Long Noncoding/genetics*
  • RNA, Long Noncoding/metabolism
  • Genetic Loci
  • Brain/metabolism
  • Craniofacial Abnormalities/genetics
  • Humans
  • Intellectual Disability/genetics
  • Heart Defects, Congenital/genetics
  • Genome
  • Introns
  • Chromosomes, Human, Pair 9/genetics
  • Mice, Inbred BALB C
  • Locomotion/genetics
  • Animals
  • Transcription Factors/metabolism
  • Zebrafish Proteins/metabolism
  • Vision, Ocular/genetics*
  • Zebrafish/embryology
  • Zebrafish/genetics
  • Zebrafish/growth & development
(all 22)
PubMed
31127312 Full text @ Nucleic Acids Res.
Abstract
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.
Genes / Markers
Figures
Figure Gallery (6 images)
Show all Figures
Expression
Gene Antibody Fish Conditions Stage Qualifier Anatomy Assay Figure
eya2ABstandard conditionsProtruding-mouthRTPCR
eya2lnc.cnib1zf3099/+standard conditionsProtruding-mouthRTPCR
foxc1aABstandard conditionsProtruding-mouthRTPCR
foxc1alnc.cnib1zf3099/+standard conditionsProtruding-mouthRTPCR
lmx1bbABstandard conditionsLong-pecISH
lmx1bbABstandard conditionsLong-pecISH
lmx1bblnc.cnib1zf3099/+standard conditionsLong-pecISH
lmx1bblnc.cnib1zf3099/+standard conditionsLong-pecISH
lnc.cnib1ABstandard conditionsProtruding-mouthISH
lnc.cnib1ABstandard conditionsProtruding-mouthISH
1 - 10 of 14
Show
Phenotype
Fish Conditions Stage Phenotype Figure
AB + MO1-lmx1bbstandard conditionsDay 5
AB + MO1-lmx1bbstandard conditionsDay 5
lnc.cnib1zf3098/+standard conditionsLong-pec
lnc.cnib1zf3098/+standard conditionsProtruding-mouth
lnc.cnib1zf3098/+standard conditionsDay 5
lnc.cnib1zf3098/+standard conditionsDay 5
lnc.cnib1zf3098/+standard conditionsDay 5
lnc.cnib1zf3098/+standard conditionsDay 5
lnc.cnib1zf3098/+standard conditionsDay 5
lnc.cnib1zf3098/+standard conditionsAdult
1 - 10 of 30
Show
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
zf3098
    		Small Deletion
    zf3099
      		Small Deletion
      1 - 2 of 2
      Show
      Human Disease / Model
      Human Disease Fish Conditions Evidence
      nail-patella syndromeTAS
      1 - 1 of 1
      Show
      Sequence Targeting Reagents
      Target Reagent Reagent Type
      lmx1bbMO1-lmx1bbMRPHLNO
      lnc.cnib1CRISPR1-lnc.cnib1CRISPR
      lnc.cnib1CRISPR2-lnc.cnib1CRISPR
      1 - 3 of 3
      Show
      Fish
      Fish
      AB
      AB + MO1-lmx1bb
      lnc.cnib1zf3098/+
      lnc.cnib1zf3098/zf3098
      lnc.cnib1zf3099/+
      1 - 5 of 5
      Show
      Antibodies
      Name Type Antigen Genes Isotypes Host Organism
      Ab3-thmonoclonalIgG1Mouse
      1 - 1 of 1
      Show
      Orthology
      No data available
      Engineered Foreign Genes
      No data available
      Mapping
      No data available
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      Citation
      Wang, F., Ren, D., Liang, X., Ke, S., Zhang, B., Hu, B., Song, X., Wang, X. (2019) A long noncoding RNA cluster-based genomic locus maintains proper development and visual function. Nucleic acids research. 47(12):6315-6329.