ZFIN ID: ZDB-FIG-191230-1549
Sucharov et al., 2019 - Selective breeding modifies mef2ca mutant incomplete penetrance by tuning the opposing Notch pathway. PLoS Genetics   15:e1008507 Full text @ PLoS Genet.
ADDITIONAL FIGURES
PHENOTYPE:
Fish:
Observed In:
Stage: Day 6

Fig 7 Loss of <italic>dlx5a</italic> function increases the penetrance of <italic>mef2ca</italic>-associated phenotypes.

(A) Animals double heterozygous for mef2ca and dlx5a were intercrossed, raised to 6 dpf and fixed and stained with Alcian Blue (cartilage) and Alizarin Red (bone). Genotyped animals were flat mounted and imaged. In this family, dlx5a homozygous mutants appear wild type. But, removing functional copies of dlx5a increases mef2ca mutant penetrance. This includes phenotypes that we interpret to be ventral-to-dorsal transformations: br-to-op transformation (arrowheads), and ch-to-hm transformation (red arrow). Scale bar: 50 μm. (B) Genotyped mef2ca;dlx5a skeletal preparations were scored for penetrance of mef2ca-associated phenotypes. Asterisks indicate significance at alpha = 0.05 Fisher’s exact test compared with dlx5a+/+. (C, D) Genotyped mef2ca;dlx5a skeletal preparations were scored for penetrance of dlx5a-associated phenotypes.

Gene Expression Details No data available
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
mef2cab1086/b1086 standard conditions Day 6 ceratohyal cartilage morphology, abnormal
Day 6 ceratohyal-interhyal joint fused with ceratohyal-interhyal joint, abnormal
Day 6 Meckel's cartilage decreased size, abnormal
Day 6 opercle mislocalised, abnormal
Day 6 quadrate-anguloarticular joint fused with quadrate-anguloarticular joint, abnormal
Day 6 symplectic decreased size, abnormal
mef2cab1086/b1086; dlx5aj1073aGt/+ standard conditions Day 6 ceratohyal cartilage morphology, exacerbated
Day 6 ceratohyal-interhyal joint fused with ceratohyal-interhyal joint, exacerbated
Day 6 Meckel's cartilage decreased size, exacerbated
Day 6 opercle mislocalised, exacerbated
Day 6 quadrate-anguloarticular joint fused with quadrate-anguloarticular joint, abnormal
Day 6 symplectic decreased size, abnormal
mef2cab1086/b1086; dlx5aj1073aGt/j1073aGt standard conditions Day 6 ceratohyal cartilage morphology, exacerbated
Day 6 ceratohyal-interhyal joint fused with ceratohyal-interhyal joint, exacerbated
Day 6 Meckel's cartilage decreased size, exacerbated
Day 6 opercle mislocalised, exacerbated
Day 6 quadrate-anguloarticular joint fused with quadrate-anguloarticular joint, abnormal
Day 6 symplectic decreased size, abnormal
Acknowledgments:
This image is the copyrighted work of the attributed author or publisher, and ZFIN has permission only to display this image to its users. Additional permissions should be obtained from the applicable author or publisher of the image. Full text @ PLoS Genet.