PUBLICATION
Involvement of the zebrafish trrap gene in craniofacial development
- Authors
- Suzuki, T., Hirai, Y., Uehara, T., Ohga, R., Kosaki, K., Kawahara, A.
- ID
- ZDB-PUB-211224-4
- Date
- 2021
- Source
- Scientific Reports 11: 24166 (Journal)
- Registered Authors
- Kawahara, Atsuo
- Keywords
- none
- MeSH Terms
-
- Adaptor Proteins, Signal Transducing*/genetics
- Adaptor Proteins, Signal Transducing*/metabolism
- Animals
- Gene Expression Regulation
- Nuclear Proteins*/genetics
- Nuclear Proteins*/metabolism
- Zebrafish*/genetics
- Zebrafish*/growth & development
- Zebrafish*/metabolism
- Zebrafish Proteins*/genetics
- Zebrafish Proteins*/metabolism
- PubMed
- 34934055 Full text @ Sci. Rep.
Citation
Suzuki, T., Hirai, Y., Uehara, T., Ohga, R., Kosaki, K., Kawahara, A. (2021) Involvement of the zebrafish trrap gene in craniofacial development. Scientific Reports. 11:24166.
Abstract
Trrap (transformation/transcription domain-associated protein) is a component shared by several histone acetyltransferase (HAT) complexes and participates in transcriptional regulation and DNA repair; however, the developmental functions of Trrap in vertebrates are not fully understood. Recently, it has been reported that human patients with genetic mutations in the TRRAP gene show various symptoms, including facial dysmorphisms, microcephaly and global developmental delay. To investigate the physiological functions of Trrap, we established trrap gene-knockout zebrafish and examined loss-of-function phenotypes in the mutants. The trrap zebrafish mutants exhibited smaller eyes and heads than the wild-type zebrafish. The size of the ventral pharyngeal arches was reduced and the mineralization of teeth was impaired in the trrap mutants. Whole-mount in situ hybridization analysis revealed that dlx3 expression was narrowly restricted in the developing ventral pharyngeal arches, while dlx2b expression was diminished in the trrap mutants. These results suggest that trrap zebrafish mutants are useful model organisms for a human disorder associated with genetic mutations in the human TRRAP gene.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping