ZFIN ID: ZDB-FIG-131210-15
Sheehan-Rooney et al., 2013 - Ahsa1 and Hsp90 activity confers more severe craniofacial phenotypes in a zebrafish model of hypoparathyroidism, sensorineural deafness and renal dysplasia (HDR). Disease models & mechanisms   6(5):1285-91 Full text @ Dis. Model. Mech.
ADDITIONAL FIGURES
EXPRESSION / LABELING:
Gene:
Antibody:
Fish:
Anatomical Terms:
Stage Range: Prim-5 to Long-pec
PHENOTYPE:
Fish:
Observed In:
Stage Range: Prim-5 to Day 5

Fig. 1

Genetic background influences the gata3 mutant phenotypes. (A–C) Flat-mounted neurocrania and (D–F) close-up views of the trabeculae (tr). In both (A) wild-type embryos and (B) mild gata3 mutants, the palate (p) is fully formed. However, there are rearrangements to the stacking of chondrocytes in mild mutants (asterisks D and E). (C,F) In severe mutants, the trabeculae are lost, generating a gap between the ethmoid plate (ep) and posterior neurocranium. (G–I) gata3 mutation disrupts outgrowth of gill buds (outlined). (H) Mild mutants generate fewer gill buds. (I) In severe mutants, the number of gill buds is further reduced. (J–L) Myosin-VI (MyoVI) labels sensory neurons (arrowheads) in the zebrafish ear. (K,L) Mild and severe mutants have fewer MyoVI -positive cells. (M–O) Wild-type and mutant embryos stained with the ret1 riboprobe. (M,N) ret1 expression is maintained in mild mutants (arrowheads). (O) In severe mutants, ret1 expression is absent. (P–R) Quantification of the defects in gata3 mutant embryos. All graphs show means ± 1 s.e.m. (P) The number of trabeculae per embryo are significantly reduced in severe mutants (average=0.17, s.e.m.=0.06, s.d.=0.46, n=58) compared with mild mutants (average=1.7, s.e.m.=0.05, s.d.=0.55, n=125). Wild-type embryos average=2 trabeculae (s.e.m.=0, s.d.=0, n=654). (Q) The number of gill buds per embryo is also significantly reduced in severe mutants (average=4.79, s.e.m.=0.57, s.d.=2.12, n=14) relative to mild mutants (average=7.92, s.e.m.=0.68, s.d.=2.35, n=12). Wild-type embryos average=12.15 gill buds (s.e.m.=0.17, s.d.=0.75, n=20). (R) Although there is a reduction compared with wild type (average=10.17, s.e.m.=0.49, s.d.=1.70, n=11), the number of MyoVI-positive cells are not significantly altered across mild (average=7.5, s.e.m.=0.27, s.d.=0.85, n=10) and severe (average=6.88, s.e.m.=0.44, s.d.=1.25, n=8) mutants. Anterior to the left; (A–F) dorsal views of flat-mounted neurocrania; (G–O) lateral views of whole-mounted embryos.

Gene Expression Details
Gene Antibody Fish Conditions Stage Qualifier Anatomy Assay
ret WT standard conditions Prim-5 pronephric distal late tubule ISH
gata3b1075/b1075(EKW) standard conditions Prim-5 Not Detected pronephric distal late tubule ISH
gata3b1075/b1075(WIK) standard conditions Prim-5 pronephric distal late tubule ISH
Antibody Labeling Details
Antibody Assay Fish Conditions Stage Anatomy
Ab1-myo6 IHC WT standard conditions Long-pec otic vesicle hair cell
IHC gata3b1075/b1075(EKW) standard conditions Long-pec otic vesicle hair cell
IHC gata3b1075/b1075(WIK) standard conditions Long-pec otic vesicle hair cell
Phenotype Details
Fish Conditions Stage Phenotype
gata3b1075/b1075(EKW) standard conditions Prim-5 pronephric distal late tubule decreased object quality, abnormal
Long-pec otic vesicle has fewer parts of type hair cell, abnormal
Day 5 embryonic neurocranium morphogenesis decreased process quality, abnormal
Day 5 internal gill bud decreased amount, abnormal
Day 5 internal gill bud decreased length, abnormal
Day 5 neurocranium lacks all parts of type neurocranial trabecula, abnormal
gata3b1075/b1075(WIK) standard conditions Long-pec otic vesicle has fewer parts of type hair cell, abnormal
Day 5 embryonic neurocranium morphogenesis decreased process quality, abnormal
Day 5 internal gill bud decreased amount, abnormal
Day 5 internal gill bud decreased length, abnormal
Day 5 neurocranial trabecula chondrocyte disorganized, abnormal
Acknowledgments:
ZFIN wishes to thank the journal Disease models & mechanisms for permission to reproduce figures from this article. Please note that this material may be protected by copyright. Full text @ Dis. Model. Mech.