PUBLICATION

Defective cranial skeletal development, larval lethality and haploinsufficiency in Myod mutant zebrafish

Authors
Hinits, Y., Williams, V.C., Sweetman, D., Donn, T.M., Ma, T.P., Moens, C.B., and Hughes, S.M.
ID
ZDB-PUB-110803-26
Date
2011
Source
Developmental Biology   358(1): 102-12 (Journal)
Registered Authors
Donn, Thomas, Hinits, Yaniv, Hughes, Simon M., Ma, Taylur, Moens, Cecilia
Keywords
muscle, zebrafish, myosin, slow, fibre, fast, myod, myogenin, myf5, miR-206, skeleton, bone, cartilage, head, fin, haploinsufficiency
MeSH Terms
  • In Situ Hybridization
  • Animals
  • Mutation/genetics
  • Cartilage/embryology
  • Haploinsufficiency/genetics*
  • Haploinsufficiency/physiology
  • Muscle Development/physiology*
  • Skull/embryology*
  • Muscle, Skeletal/embryology
  • Upper Extremity/embryology
  • MyoD Protein/genetics*
  • MyoD Protein/metabolism
  • Gene Expression Regulation, Developmental/physiology*
  • Bone and Bones/embryology*
  • Immunohistochemistry
  • Larva/physiology
  • Zebrafish/embryology*
  • Zebrafish/genetics
(all 18)
PubMed
21798255 Full text @ Dev. Biol.
Abstract
Myogenic regulatory factors of the myod family (MRFs) are transcription factors essential for mammalian skeletal myogenesis. Here we show that a mutation in the zebrafish myod gene delays and reduces early somitic and pectoral fin myogenesis, reduces miR-206 expression, and leads to a persistent reduction in somite size until at least the independent feeding stage. A mutation in myog, encoding a second MRF, has little obvious phenotype at early stages, but exacerbates the loss of somitic muscle caused by lack of Myod. Mutation of both myod and myf5 ablates all skeletal muscle. Haploinsufficiency of myod leads to reduced embryonic somite muscle bulk. Lack of Myod causes a severe reduction in cranial musculature, ablating most muscles including the protractor pectoralis, a putative cucullaris homologue. This phenotype is accompanied by a severe dysmorphology of the cartilaginous skeleton and failure of maturation of several cranial bones, including the opercle. As myod expression is restricted to myogenic cells, the data show that myogenesis is essential for proper skeletogenesis in the head.
Genes / Markers
Figures
Figure Gallery (9 images)
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Expression
Phenotype
No data available
Mutations / Transgenics
Allele Construct Type Affected Genomic Region
fh261
    Point Mutation
    fh265
      Point Mutation
      hu2022
        Point Mutation
        i135TgTransgenic Insertion
          vu119TgTransgenic Insertion
            zf13TgTransgenic Insertion
              1 - 6 of 6
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              Human Disease / Model
              No data available
              Sequence Targeting Reagents
              Target Reagent Reagent Type
              myod1MO1-myod1MRPHLNO
              1 - 1 of 1
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              Fish
              Antibodies
              Orthology
              No data available
              Engineered Foreign Genes
              Marker Marker Type Name
              EGFPEFGEGFP
              GFPEFGGFP
              1 - 2 of 2
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              Mapping
              No data available