PUBLICATION

Foxp- and Skor-family proteins control differentiation of Purkinje cells from Ptf1a and Neurogenin1-expressing progenitors in zebrafish

Authors
Itoh, T., Uehara, M., Yura, S., Wang, J.C., Fujii, Y., Nakanishi, A., Shimizu, T., Hibi, M.
ID
ZDB-PUB-240309-2
Date
2024
Source
Development (Cambridge, England)   151(7): (Journal)
Registered Authors
Hibi, Masahiko, Shimizu, Takashi
Keywords
Foxp, Neurogenin1, Ptf1a, Purkinje cells, Skor, Zebrafish
MeSH Terms
  • Neurons/metabolism
  • Cell Differentiation/genetics
  • Purkinje Cells*/metabolism
  • Zebrafish*/genetics
  • Mammals
  • Animals
  • Cerebellum
(all 7)
PubMed
38456494 Full text @ Development
Abstract
Cerebellar neurons, such as GABAergic Purkinje cells (PCs), interneurons (INs), and glutamatergic granule cells (GCs) are differentiated from neural progenitors expressing proneural genes including ptf1a, neurogenin1, and atoh1a/b/c. Studies in mammals previously suggested that these genes determine cerebellar neuron cell fate. However, our studies on ptf1a;neurogenin1 zebrafish mutants and lineage tracing of ptf1a-expressing progenitors have revealed that the ptf1a/neurogenin1-expressing progenitors can generate diverse cerebellar neurons including PCs, INs, and a part of GCs in zebrafish. The precise mechanisms of how each cerebellar neuron type is specified remains elusive. We found that genes encoding transcriptional regulators Foxp1b, Foxp4, Skor1b, and Skor2, which are reportedly expressed in PCs, were absent in ptf1a;neurogenin1 mutants. foxp1b;foxp4 mutants showed a strong reduction in PCs, while skor1b;skor2 mutants completely lacked PCs but instead displayed an increase in immature GCs. Misexpression of skor2 in GC progenitors expressing atoh1c suppressed GC fate. These data indicate that Foxp1b/4 and Skor1b/2 function as key transcriptional regulators in the initial step of PC differentiation from ptf1a/neurogenin1-expressing neural progenitors, while Skor1b and Skor2 control PC differentiation by suppressing their differentiation into GCs.
Genes / Markers
Figures
Figure Gallery (10 images)
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Expression
No data available
Phenotype
No data available
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
fh430TgTransgenic Insertion
    hi1059TgTransgenic Insertion
    jh1TgTransgenic Insertion
      jh16TgTransgenic Insertion
        nkuasrfp1aTgTransgenic Insertion
          nns14TgTransgenic Insertion
            nns26TgTransgenic Insertion
              nns27TgTransgenic Insertion
                nub34
                  		Small Deletion
                  nub89
                    		Small Deletion
                    1 - 10 of 21
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                    Human Disease / Model
                    No data available
                    Sequence Targeting Reagents
                    Target Reagent Reagent Type
                    foxp1bCRISPR1-foxp1bCRISPR
                    foxp1bCRISPR2-foxp1bCRISPR
                    foxp4CRISPR1-foxp4CRISPR
                    foxp4CRISPR2-foxp4CRISPR
                    lhx1aCRISPR1-lhx1aCRISPR
                    lhx1bCRISPR1-lhx1bCRISPR
                    lhx5CRISPR1-lhx5CRISPR
                    skor1bCRISPR1-skor1bCRISPR
                    skor1bCRISPR2-skor1bCRISPR
                    skor2CRISPR1-skor2CRISPR
                    1 - 10 of 11
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                    Fish
                    No data available
                    Antibodies
                    Name Type Antigen Genes Isotypes Host Organism
                    Ab1-aldocmonoclonalMouse
                    Ab1-neurodmonoclonalMouse
                    Ab1-pvalb7monoclonalMouse
                    Ab1-slc17a7polyclonalRabbit
                    Ab2-ca8monoclonalMouse
                    1 - 5 of 5
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                    Orthology
                    No data available
                    Engineered Foreign Genes
                    Marker Marker Type Name
                    TagCFPEFGTagCFP
                    CreEFGCre
                    DsRedEFGDsRed
                    EGFPEFGEGFP
                    GAL4EFGGAL4
                    GAL4FFEFGGAL4FF
                    GFPEFGGFP
                    KaedeEFGKaede
                    mCherryEFGmCherry
                    RFPEFGRFP
                    1 - 10 of 10
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                    Mapping
                    No data available
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                    Citation
                    Itoh, T., Uehara, M., Yura, S., Wang, J.C., Fujii, Y., Nakanishi, A., Shimizu, T., Hibi, M. (2024) Foxp- and Skor-family proteins control differentiation of Purkinje cells from Ptf1a and Neurogenin1-expressing progenitors in zebrafish. Development (Cambridge, England). 151(7):.