PUBLICATION
Identification of adult mineralized tissue zebrafish mutants
- Authors
- Andreeva, V., Connolly, M.H., Stewart-Swift, C., Fraher, D., Burt, J., Cardarelli, J., and Yelick, P.C.
- ID
- ZDB-PUB-110119-23
- Date
- 2011
- Source
- Genesis (New York, N.Y. : 2000) 49(4): 360-366 (Journal)
- Registered Authors
- Burt, Jeff, Yelick, Pamela C.
- Keywords
- adult craniofacial, skeletal, tooth dysplasias
- MeSH Terms
-
- Cartilage/abnormalities*
- Animals
- Alcian Blue
- Craniofacial Abnormalities/genetics*
- Genes, Recessive/genetics
- Ethylnitrosourea
- Phenotype*
- Zebrafish
- Anthraquinones
- Mutagenesis
- Disease Models, Animal*
- PubMed
- 21225658 Full text @ Genesis
Citation
Andreeva, V., Connolly, M.H., Stewart-Swift, C., Fraher, D., Burt, J., Cardarelli, J., and Yelick, P.C. (2011) Identification of adult mineralized tissue zebrafish mutants. Genesis (New York, N.Y. : 2000). 49(4):360-366.
Abstract
Zebrafish craniofacial, skeletal, and tooth development closely resembles that of higher vertebrates. Our goal is to identify viable adult zebrafish mutants that can be used as models for human mineralized craniofacial, dental, and skeletal system disorders. We utilized a large-scale forward-genetic chemical N-ethyl-nitroso-urea (ENU) mutagenesis screen to identify 17 early lethal homozygous recessive mutants with defects in craniofacial cartilage elements, and 7 adult homozygous recessive mutants with mineralized tissue phenotypes including craniofacial shape defects, fused sutures, dysmorphic or missing skeletal elements, scoliosis, and neural arch defects. One mutant displayed both an early lethal homozygous phenotype and an adult heterozygous phenotype. These results extend the utility of the zebrafish model beyond the embryo, to study human bone and cartilage disorders.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping