PUBLICATION

Abrogation of Stem Loop Binding Protein (Slbp) function leads to a failure of cells to transition from proliferation to differentiation, retinal coloboma and midline axon guidance deficits

Authors
Turner, K.J., Hoyle, J., Valdivia, L.E., Cerveny, K.L., Hart, W., Mangoli, M., Geisler, R., Rees, M., Houart, C., Poole, R.J., Wilson, S.W., Gestri, G.
ID
ZDB-PUB-190130-16
Date
2019
Source
PLoS One   14: e0211073 (Journal)
Registered Authors
Cerveny, Kara, Geisler, Robert, Gestri, Gaia, Houart, Corinne, Hoyle, Jacqueline, Mangoli, Maryam, Poole, Richard, Rees, Michele, Turner, Katherine, Valdivia, Leonardo, Wilson, Steve
Keywords
none
MeSH Terms
  • Animals
  • Animals, Genetically Modified*/embryology
  • Animals, Genetically Modified*/genetics
  • Axon Guidance/genetics*
  • Cell Differentiation*
  • Cell Proliferation*
  • Coloboma*/embryology
  • Coloboma*/genetics
  • Coloboma*/pathology
  • Histones/genetics
  • Histones/metabolism
  • RNA, Messenger/biosynthesis
  • RNA, Messenger/genetics
  • Retinal Diseases*/embryology
  • Retinal Diseases*/genetics
  • Retinal Diseases*/pathology
  • Zebrafish*/embryology
  • Zebrafish*/genetics
  • Zebrafish Proteins/deficiency*
(all 19)
PubMed
30695021 Full text @ PLoS One
CTD
30695021
Abstract
Through forward genetic screening for mutations affecting visual system development, we identified prominent coloboma and cell-autonomous retinal neuron differentiation, lamination and retinal axon projection defects in eisspalte (ele) mutant zebrafish. Additional axonal deficits were present, most notably at midline axon commissures. Genetic mapping and cloning of the ele mutation showed that the affected gene is slbp, which encodes a conserved RNA stem-loop binding protein involved in replication dependent histone mRNA metabolism. Cells throughout the central nervous system remained in the cell cycle in ele mutant embryos at stages when, and locations where, post-mitotic cells have differentiated in wild-type siblings. Indeed, RNAseq analysis showed down-regulation of many genes associated with neuronal differentiation. This was coincident with changes in the levels and spatial localisation of expression of various genes implicated, for instance, in axon guidance, that likely underlie specific ele phenotypes. These results suggest that many of the cell and tissue specific phenotypes in ele mutant embryos are secondary to altered expression of modules of developmental regulatory genes that characterise, or promote transitions in, cell state and require the correct function of Slbp-dependent histone and chromatin regulatory genes.
Genes / Markers
Marker Marker Type Name
actb1GENEactin, beta 1
aldh1a3GENEaldehyde dehydrogenase 1 family, member A3
ccnd1GENEcyclin D1
ccne1GENEcyclin E1
cdt1GENEchromatin licensing and DNA replication factor 1
chd7GENEchromodomain helicase DNA binding protein 7
col10a1aGENEcollagen, type X, alpha 1a
crabp1bGENEcellular retinoic acid binding protein 1b
elavl2GENEELAV like neuron-specific RNA binding protein 2
gmnnGENEgeminin DNA replication inhibitor
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Figures
Figure Gallery (9 images)
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Expression
Phenotype
Fish Conditions Stage Phenotype Figure
WT + MO1-slbpstandard conditionsPrim-15
WT + MO1-slbpstandard conditionsPrim-15
WT + MO1-slbpstandard conditionsPrim-15
WT + MO1-slbpstandard conditionsPrim-15
WT + MO1-slbpstandard conditionsPrim-15
WT + MO1-slbpstandard conditionsPrim-15
WT + MO1-slbpstandard conditionsPrim-15
WT + MO1-slbpstandard conditionsPrim-15
WT + MO1-slbpstandard conditionsLong-pec
WT + MO1-slbpstandard conditionsLong-pec
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Mutations / Transgenics
Allele Construct Type Affected Genomic Region
b1205TgTransgenic Insertion
    rw021TgTransgenic Insertion
      rw0405bTgTransgenic Insertion
        rw0410hTgTransgenic Insertion
          sb1TgTransgenic Insertion
            ty77e
              		Point Mutation
              1 - 6 of 6
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              Human Disease / Model
              No data available
              Sequence Targeting Reagents
              Target Reagent Reagent Type
              slbpMO1-slbpMRPHLNO
              1 - 1 of 1
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              Fish
              Fish
              b1205Tg
              rw021Tg
              rw0405bTg; rw0410hTg
              sb1Tg
              slbpty77e/ty77e
              slbpty77e/ty77e; b1205Tg
              slbpty77e/ty77e; rw021Tg
              slbpty77e/ty77e; rw0405bTg; rw0410hTg
              slbpty77e/ty77e; sb1Tg
              WT
              1 - 10 of 11
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              Antibodies
              Orthology
              No data available
              Engineered Foreign Genes
              Marker Marker Type Name
              GFPEFGGFP
              mAGFPEFGmAGFP
              mKOFP2EFGmKOFP2
              1 - 3 of 3
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              Mapping
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              Citation
              Turner, K.J., Hoyle, J., Valdivia, L.E., Cerveny, K.L., Hart, W., Mangoli, M., Geisler, R., Rees, M., Houart, C., Poole, R.J., Wilson, S.W., Gestri, G. (2019) Abrogation of Stem Loop Binding Protein (Slbp) function leads to a failure of cells to transition from proliferation to differentiation, retinal coloboma and midline axon guidance deficits. PLoS One. 14:e0211073.