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
  • Animals
  • Bone and Bones/embryology*
  • Cartilage/embryology
  • Gene Expression Regulation, Developmental/physiology*
  • Haploinsufficiency/genetics*
  • Haploinsufficiency/physiology
  • Immunohistochemistry
  • In Situ Hybridization
  • Larva/physiology
  • Muscle Development/physiology*
  • Muscle, Skeletal/embryology
  • Mutation/genetics
  • MyoD Protein/genetics*
  • MyoD Protein/metabolism
  • Skull/embryology*
  • Upper Extremity/embryology
  • Zebrafish/embryology*
  • Zebrafish/genetics
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
Expression
Phenotype
Mutation and Transgenics
Human Disease / Model Data
Sequence Targeting Reagents
Fish
Antibodies
Orthology
Engineered Foreign Genes
Mapping
Errata and Notes