ZFIN ID: ZDB-FIG-210216-2
Castro et al., 2020 - Activation of WNT signaling restores the facial deficits in a zebrafish with defects in cholesterol metabolism. Genesis (New York, N.Y. : 2000)   58(12):e23397 Full text @ Genesis
ADDITIONAL FIGURES
EXPRESSION / LABELING:
Genes:
Fish:
Condition:
Anatomical Term:
Stage: Long-pec
PHENOTYPE:
Fish:
Condition:
Observed In:
Stage Range: Long-pec to Day 4

Fig. 2 Activation of WNT signaling restores the facial defects present in the Vu57 allele. (a) Experimental design schematic with onset of treatment with WNT‐Agonist I at 30 hours post fertilization (HPF) and removal of treatment at 54 HPF. Alcian Blue was performed at 4 days post fertilization (DPF) in treated and untreated individuals that were incubated in embryo media from 54–96 HPF following treatment. (b–e) Alcian blue staining was performed at 4 DPF in homozygous carriers of the Vu57 allele (Vu57−/−) and wild type siblings (Sibling) according to the treatment schematic in (a). Total numbers of animals reflected in Table 1. *p = .0001 using a Fisher's Exact test. (f). Homozygous carriers of the Vu57 allele (Vu57−/−) or wild type siblings (Sibling) were treated with vehicle control (DMSO) or WNT‐agonist I at either 0.1 or 0.2 μM concentration according to the schematic in (a). Total RNA was isolated from a pool of embryos and the expression of sox10, col2a1a, or axin2 was measured by quantitative PCR (qPCR). Error bars represent SD of biological replicates. N = 11/group except for 0.2 μM concentration where N = 9. *p = 4.07457E‐05, **1.14579E‐06, ***p = 3.80572E‐07, #2.45716E‐05, ##p = 7.28315E‐08, ###p = .000716073, &p = 3.22846E‐05, &&p = 7.2866E‐07, &&&p = 8.45279E‐07

Gene Expression Details
Gene Antibody Fish Conditions Stage Anatomy Assay
axin2 hmgcs1vu57/vu57 standard conditions Long-pec whole organism RTPCR
hmgcs1vu57/vu57 chemical treatment by environment: Wnt signalling activator Long-pec whole organism RTPCR
col2a1a hmgcs1vu57/vu57 standard conditions Long-pec whole organism RTPCR
hmgcs1vu57/vu57 chemical treatment by environment: Wnt signalling activator Long-pec whole organism RTPCR
sox10 hmgcs1vu57/vu57 standard conditions Long-pec whole organism RTPCR
hmgcs1vu57/vu57 chemical treatment by environment: Wnt signalling activator Long-pec whole organism RTPCR
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
hmgcs1vu57/vu57 standard conditions Long-pec whole organism axin2 expression decreased amount, abnormal
Long-pec whole organism col2a1a expression decreased amount, abnormal
Long-pec whole organism sox10 expression decreased amount, abnormal
Day 4 ceratobranchial cartilage absent, abnormal
Day 4 ceratohyal cartilage absent, abnormal
Day 4 embryonic viscerocranium morphogenesis disrupted, abnormal
Day 4 Meckel's cartilage absent, abnormal
hmgcs1vu57/vu57 chemical treatment by environment: Wnt signalling activator Long-pec whole organism axin2 expression amount, ameliorated
Long-pec whole organism col2a1a expression amount, ameliorated
Long-pec whole organism sox10 expression amount, ameliorated
Day 4 ceratobranchial cartilage present, ameliorated
Day 4 ceratohyal cartilage present, ameliorated
Day 4 embryonic viscerocranium morphogenesis process quality, ameliorated
Day 4 Meckel's cartilage present, ameliorated
Acknowledgments:
ZFIN wishes to thank the journal Genesis (New York, N.Y. : 2000) for permission to reproduce figures from this article. Please note that this material may be protected by copyright. Full text @ Genesis