ZFIN ID: ZDB-FIG-191230-1553
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
EXPRESSION / LABELING:
Genes:
Fish:
Anatomical Terms:
Stage: Long-pec
PHENOTYPE:
Fish:
Observed In:
Stage Range: Long-pec to Day 6

Fig 11 Triple mutants reveal <italic>mef2ca</italic> modifier epistasis.

(A) Animals triple heterozygous for mef2ca, dlx5a and jag1b were intercrossed, offspring were raised to 6 dpf and fixed and stained with Alcian Blue (cartilage) and Alizarin Red (bone). Genotyped skeletal preparations were scored for penetrance of mef2ca-associated phenotypes. Removing functional copies of jag1b from mef2ca;dlx5a double mutants decreases penetrance of mef2ca-associated phenotypes. Asterisks indicate significance at alpha = 0.05 Fisher’s exact test compared with mef2ca-/-;dlx5a+/+;jag1b+/+. (B) Genotyped mef2ca;dlx5a;jag1b skeletal preparations were flat mounted and imaged. mef2ca mutant associated phenotypes are indicated including ectopic bone (arrowheads), dysmorphic ch (arrows) and ch-to-hm transformation (red arrow). Stick-like opercle bone is indicated with a white arrow. Scale bar: 50 μm. (C) Head expression of Notch target genes was quantified by RT-qPCR in full-sibling dlx5a mutant and wild-type embryos at 48 hpf. Expression in dlx5a mutants was normalized to wild-type sibling expression levels (black line). Asterisks mark genes with expression levels significantly different in dlx5a mutants compared with dlx5a wild types (T-test at alpha = 0.05). Error bars are standard deviation. (D) Spatial her6 expression was monitored by in situ hybridization in wild types and dlx5a mutants. In wild-type embryos her6 is expressed in three discrete patches in arch 2 (arrows). In dlx5a homozygous mutants, her6 expands ventrally resulting in a larger, more continuous expression domain (arrowhead). dlx2a expression was used to delineate pharyngeal arches one (a1) and two (a2) (dashed outlines). her6 was expanded in 0/4 homozygous wild types and 3/3 homozygous mutants Scale bar: 30 μm.

Gene Expression Details
Gene Antibody Fish Conditions Stage Anatomy Assay
dlx2a WT standard conditions Long-pec pharyngeal arch 1 ISH
Long-pec pharyngeal arch 2 ISH
dlx5aj1073aGt/j1073aGt standard conditions Long-pec pharyngeal arch 1 ISH
Long-pec pharyngeal arch 2 ISH
her6 WT standard conditions Long-pec pharyngeal arch 2 ISH
dlx5aj1073aGt/j1073aGt standard conditions Long-pec head RTPCR
Long-pec pharyngeal arch 2 ISH
hey1 dlx5aj1073aGt/j1073aGt standard conditions Long-pec head RTPCR
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
dlx5aj1073aGt/j1073aGt standard conditions Long-pec head her6 expression increased amount, abnormal
Long-pec pharyngeal arch 2 her6 expression increased distribution, abnormal
jag1bb1105/+; mef2cab1086/b1086; dlx5aj1073aGt/j1073aGt standard conditions Day 6 ceratohyal cartilage morphology, ameliorated
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
jag1bb1105/b1105; mef2cab1086/b1086; dlx5aj1073aGt/j1073aGt standard conditions Day 6 ceratohyal cartilage morphology, ameliorated
Day 6 ceratohyal-interhyal joint fused with ceratohyal-interhyal joint, abnormal
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
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, abnormal
Day 6 ceratohyal-interhyal joint fused with ceratohyal-interhyal joint, abnormal
Day 6 Meckel's cartilage decreased size, abnormal
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, abnormal
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.