PUBLICATION
Comprehensive series of Irx cluster mutants reveals diverse roles in facial cartilage development
- Authors
- Farmer, D.T., Patel, P., Choi, R., Liu, C.Y., Crump, J.G.
- ID
- ZDB-PUB-210803-8
- Date
- 2021
- Source
- Development (Cambridge, England) 148(16): (Journal)
- Registered Authors
- Keywords
- Cartilage, Craniofacial skeleton, Fin, Irx, Zebrafish
- MeSH Terms
-
- Alleles
- Animals
- Animals, Genetically Modified
- Body Patterning/genetics
- Cartilage/embryology*
- Chondrogenesis/genetics*
- Gene Expression
- Gene Expression Regulation, Developmental
- Homeodomain Proteins/genetics
- Homeodomain Proteins/metabolism*
- Multigene Family*
- Mutant Proteins/metabolism*
- Mutation
- Neural Crest/metabolism
- Skull/embryology*
- Transcription Factors/genetics
- Transcription Factors/metabolism*
- Zebrafish/embryology*
- Zebrafish/genetics*
- Zebrafish Proteins/genetics
- Zebrafish Proteins/metabolism*
- PubMed
- 34338288 Full text @ Development
Citation
Farmer, D.T., Patel, P., Choi, R., Liu, C.Y., Crump, J.G. (2021) Comprehensive series of Irx cluster mutants reveals diverse roles in facial cartilage development. Development (Cambridge, England). 148(16).
Abstract
Proper function of the vertebrate skeleton requires the development of distinct articulating embryonic cartilages. Irx transcription factors are arranged in co-regulated clusters that are expressed in the developing skeletons of the face and appendages. IrxB cluster genes are required for the separation of toes in mice and formation of the hyoid joint in zebrafish, yet whether Irx genes had broader roles in skeletal development remained unclear. Here we perform a comprehensive loss-of-function analysis of all 11 Irx genes in zebrafish. We uncover conserved requirements for IrxB genes in formation of the fish and mouse scapula. In the face, we find a requirement for IrxAb genes and irx7 in formation of anterior neural crest precursors of the jaw, and for IrxBa genes in formation of endodermal pouches and gill cartilages. We also observe extensive joint loss and cartilage fusions in animals with combinatorial losses of Irx clusters, with in vivo imaging revealing that at least some of these fusions arise through inappropriate chondrogenesis. Our analysis reveals diverse roles for Irx genes in the formation and later segmentation of the facial skeleton.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping