PUBLICATION
Generation and characterization of Kctd15 mutations in zebrafish
- Authors
- Heffer, A., Marquart, G.D., Aquilina-Beck, A., Saleem, N., Burgess, H.A., Dawid, I.B.
- ID
- ZDB-PUB-171208-8
- Date
- 2017
- Source
- PLoS One 12: e0189162 (Journal)
- Registered Authors
- Burgess, Harold, Dawid, Igor B., Marquart, Gregory
- Keywords
- none
- MeSH Terms
-
- Animals
- Frameshift Mutation*
- Potassium Channels, Voltage-Gated/genetics*
- Zebrafish/genetics*
- Zebrafish Proteins/genetics*
- PubMed
- 29216270 Full text @ PLoS One
Citation
Heffer, A., Marquart, G.D., Aquilina-Beck, A., Saleem, N., Burgess, H.A., Dawid, I.B. (2017) Generation and characterization of Kctd15 mutations in zebrafish. PLoS One. 12:e0189162.
Abstract
Potassium channel tetramerization domain containing 15 (Kctd15) was previously found to have a role in early neural crest (NC) patterning, specifically delimiting the region where NC markers are expressed via repression of transcription factor AP-2a and inhibition of Wnt signaling. We used transcription activator-like effector nucleases (TALENs) to generate null mutations in zebrafish kctd15a and kctd15b paralogs to study the in vivo role of Kctd15. We found that while deletions producing frame-shift mutations in each paralog showed no apparent phenotype, kctd15a/b double mutant zebrafish are smaller in size and show several phenotypes including some affecting the NC, such as expansion of the early NC domain, increased pigmentation, and craniofacial defects. Both melanophore and xanthophore pigment cell numbers and early markers are up-regulated in the double mutants. While we find no embryonic craniofacial defects, adult mutants have a deformed maxillary segment and missing barbels. By confocal imaging of mutant larval brains we found that the torus lateralis (TLa), a region implicated in gustatory networks in other fish, is absent. Ablation of this brain tissue in wild type larvae mimics some aspects of the mutant growth phenotype. Thus kctd15 mutants show deficits in the development of both neural crest derivatives, and specific regions within the central nervous system, leading to a strong reduction in normal growth rates.
Genes / Markers
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping