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
-
- Larva/physiology
- Animals
- Bone and Bones/embryology*
- MyoD Protein/genetics*
- MyoD Protein/metabolism
- Zebrafish/embryology*
- Zebrafish/genetics
- Muscle Development/physiology*
- Cartilage/embryology
- Immunohistochemistry
- Mutation/genetics
- Haploinsufficiency/genetics*
- Haploinsufficiency/physiology
- Skull/embryology*
- Gene Expression Regulation, Developmental/physiology*
- Muscle, Skeletal/embryology
- In Situ Hybridization
- Upper Extremity/embryology
- PubMed
- 21798255 Full text @ Dev. Biol.
Citation
Hinits, Y., Williams, V.C., Sweetman, D., Donn, T.M., Ma, T.P., Moens, C.B., and Hughes, S.M. (2011) Defective cranial skeletal development, larval lethality and haploinsufficiency in Myod mutant zebrafish. Developmental Biology. 358(1):102-12.
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
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping