ZFIN ID: ZDB-FIG-191230-1550
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:
Condition:
Observed In:
Stage: Day 6

Fig 8 Pharmacological Notch inhibition decreases the penetrance of <italic>mef2ca</italic>-associated phenotypes without producing a skeletal phenotype in genetic wild types.

(A) Animals heterozygous for mef2ca from the high-penetrance strain were intercrossed and offspring treated with Notch inhibitor (DBZ) or vehicle control (DMSO) from 18–48 hpf. Treated animals were fixed and stained with Alcian Blue (cartilage) and Alizarin Red (bone) at 6 dpf. Genotyped skeletal preparations were scored for penetrance of mef2ca-associated phenotypes. Asterisks indicate significance at alpha = 0.05 Fisher’s exact test compared with DMSO. (B) Imaged flat mounts of dissected skeletons from stained, genotyped animals treated with DMSO or DBZ were imaged. Arrows indicate dysmorphic ch and arrowheads mark ectopic bone. Scale bar: 50 μm. (C) Animals heterozygous for mef2ca from an unselected strain were intercrossed and offspring treated with either DMSO from 18–48 hpf, or Notch inhibitor (DBZ) from 18–30, or DBZ from 30–48 hpf. Treated animals were fixed and stained with Alcian Blue (cartilage) and Alizarin Red (bone) at 6 dpf. Genotyped skeletal preparations were scored for penetrance of mef2ca-associated phenotypes. Asterisks indicate significance at alpha = 0.05 Fisher’s exact test compared with DMSO.

Gene Expression Details No data available
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
mef2cab1086/b1086 control 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 chemical treatment: dibenzoazepine Day 6 ceratohyal cartilage morphology, ameliorated
Day 6 ceratohyal-interhyal joint fused with ceratohyal-interhyal joint, ameliorated
Day 6 Meckel's cartilage decreased size, abnormal
Day 6 opercle mislocalised, ameliorated
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.