ZFIN ID: ZDB-FIG-180608-108
Barske et al., 2018 - Essential Role of Nr2f Nuclear Receptors in Patterning the Vertebrate Upper Jaw. Developmental Cell   44(3):337-347.e5 Full text @ Dev. Cell
ADDITIONAL FIGURES
EXPRESSION / LABELING:
Genes:
Fish:
Anatomical Terms:
Stage: Prim-25
PHENOTYPE:
Fish:
Observed In:
Stage Range: Prim-25 to Day 5

Fig. 6

Reducing Nr2f Gene Dosage Can Fully Rescue the Lower Jaw in edn1 Mutants

(A–E) Facial skeletons of fish carrying nr2f and/or edn1 mutations, schematized in (A′–E′). Compared to the control (A), nr2f2; nr2f5 mutants (B) have a thickened upper jaw (double arrowhead), while edn1 mutants (C) have a much reduced lower jaw. Reducing Nr2f alleles in edn1 mutants (D and E) can rescue the jaw joint (black arrows) and Meckel's cartilage (compare C with D–E). The upper jaw is still enlarged in nr2f2; nr2f5; edn1 combinatorial mutants (E, double arrowhead). M, Meckel's cartilage; Ptp, pterygoid process; jj, jaw joint. Scale bar in (E) 50 μm.

(F and G) Quantification of jaw joint (F) and Meckel's rescue (G) across different summed nr2f; edn1 genotypes. One or both jaw joints were rescued in 12/106 mutants lacking three to six Nr2f alleles. See Figure S6 for Meckel's scoring system. n = 16, 29, 30, 53, 42, 10, and 2 for edn1 mutants with zero to six mutant Nr2f alleles, respectively. Error bars are the SEM. Scores in (G) were non-evenly distributed across genotype classes (p < 0.0001, chi-squared).

(H–J) dlx5a (H), dlx6a (I), and hand2 (J) (magenta) are expanded in nr2f2; nr2f5 mutants (white arrowheads), reduced in edn1 mutants, and restored in the first arch of nr2f5; edn1 mutants (nr2f1b/nr2f2 genotypes indicated). Some ectopic expansion into maxillary cells was still evident in the combinatorial mutants (white arrowheads), yet second-arch expression was not restored (double arrowheads). dlx2a (green) labels arch NCCs, and dashed lines in (J) indicate arch boundaries.

(K) Ectopic ventral expression of jag1b (magenta) and prrx1b (green) in edn1 mutants (white arrows) is reversed in the mandibular (asterisk) but not hyoid (double arrowheads) domains of nr2f1b; nr2f2; nr2f5; edn1 mutants. White outlines depict the first endodermal pouch.

Scale bars in (E) 50 μm, (H–K) 20 μm.

Gene Expression Details
Gene Antibody Fish Conditions Stage Anatomy Assay
dlx2a TU standard conditions Prim-25 pharyngeal arch 1 neural crest cell ISH
Prim-25 pharyngeal arch 2 neural crest cell ISH
edn1tf216b/tf216b (TU) standard conditions Prim-25 pharyngeal arch 1 neural crest cell ISH
Prim-25 pharyngeal arch 2 neural crest cell ISH
edn1tf216b/tf216b; nr2f1bel523/el523; nr2f2el506/+; nr2f5el611/el611 (TU) standard conditions Prim-25 pharyngeal arch 1 neural crest cell ISH
Prim-25 pharyngeal arch 2 neural crest cell ISH
nr2f1bel523/el523; nr2f2el506/el506; nr2f5el611/el611 (TU) standard conditions Prim-25 pharyngeal arch 1 neural crest cell ISH
Prim-25 pharyngeal arch 2 neural crest cell ISH
nr2f2el506/el506; nr2f5el611/el611 (TU) standard conditions Prim-25 pharyngeal arch 1 neural crest cell ISH
Prim-25 pharyngeal arch 2 neural crest cell ISH
dlx5a TU standard conditions Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
edn1tf216b/tf216b (TU) standard conditions Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
edn1tf216b/tf216b; nr2f1bel523/el523; nr2f2el506/+; nr2f5el611/el611 (TU) standard conditions Prim-25 maxilla ISH
Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
nr2f1bel523/el523; nr2f2el506/el506; nr2f5el611/el611 (TU) standard conditions Prim-25 maxilla ISH
Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
dlx6a TU standard conditions Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
edn1tf216b/tf216b (TU) standard conditions Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
edn1tf216b/tf216b; nr2f1bel523/el523; nr2f2el506/+; nr2f5el611/el611 (TU) standard conditions Prim-25 maxilla ISH
Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
nr2f2el506/el506; nr2f5el611/el611 (TU) standard conditions Prim-25 maxilla ISH
Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
hand2 TU standard conditions Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
edn1tf216b/tf216b (TU) standard conditions Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
edn1tf216b/tf216b; nr2f1bel523/+; nr2f2el506/el506; nr2f5el611/el611 (TU) standard conditions Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
nr2f1bel523/+; nr2f2el506/el506; nr2f5el611/el611 (TU) standard conditions Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
jag1b TU standard conditions Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
edn1tf216b/tf216b (TU) standard conditions Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
edn1tf216b/tf216b; nr2f1bel523/el523; nr2f2el506/el506; nr2f5el611/el611 (TU) standard conditions Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
nr2f1bel523/el523; nr2f2el506/el506; nr2f5el611/el611 (TU) standard conditions Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
prrx1b TU standard conditions Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
edn1tf216b/tf216b (TU) standard conditions Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
edn1tf216b/tf216b; nr2f1bel523/el523; nr2f2el506/el506; nr2f5el611/el611 (TU) standard conditions Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
nr2f1bel523/el523; nr2f2el506/el506; nr2f5el611/el611 (TU) standard conditions Prim-25 pharyngeal arch 1 ISH
Prim-25 pharyngeal arch 2 ISH
Antibody Labeling Details No data available
Phenotype Details
Fish Conditions Stage Phenotype
edn1tf216b/tf216b (TU) standard conditions Prim-25 pharyngeal arch 1 dlx5a expression decreased amount, abnormal
Prim-25 pharyngeal arch 1 dlx6a expression decreased amount, abnormal
Prim-25 pharyngeal arch 1 hand2 expression decreased amount, abnormal
Prim-25 pharyngeal arch 1 jag1b expression increased distribution, abnormal
Prim-25 pharyngeal arch 1 prrx1b expression increased distribution, abnormal
Prim-25 pharyngeal arch 2 dlx5a expression decreased amount, abnormal
Prim-25 pharyngeal arch 2 dlx6a expression decreased amount, abnormal
Prim-25 pharyngeal arch 2 hand2 expression decreased amount, abnormal
Prim-25 pharyngeal arch 2 jag1b expression increased distribution, abnormal
Prim-25 pharyngeal arch 2 prrx1b expression increased distribution, abnormal
Day 5 Meckel's cartilage absent, abnormal
Day 5 quadrate-anguloarticular joint absent, abnormal
Day 5 ventral mandibular arch decreased size, abnormal
edn1tf216b/tf216b; nr2f1bel523/+; nr2f2el506/el506; nr2f5el611/el611 (TU) standard conditions Prim-25 pharyngeal arch 1 hand2 expression spatial pattern, ameliorated
Prim-25 pharyngeal arch 2 hand2 expression spatial pattern, ameliorated
edn1tf216b/tf216b; nr2f1bel523/el523; nr2f2el506/+; nr2f5el611/el611 (TU) standard conditions Prim-25 maxilla dlx5a expression increased amount, abnormal
Prim-25 maxilla dlx6a expression increased amount, abnormal
Prim-25 pharyngeal arch 1 dlx5a expression spatial pattern, ameliorated
Prim-25 pharyngeal arch 1 dlx6a expression spatial pattern, ameliorated
Prim-25 pharyngeal arch 2 dlx5a expression increased distribution, abnormal
Prim-25 pharyngeal arch 2 dlx6a expression increased distribution, abnormal
Day 5 Meckel's cartilage morphology, ameliorated
Day 5 quadrate-anguloarticular joint morphology, ameliorated
edn1tf216b/tf216b; nr2f1bel523/el523; nr2f2el506/el506; nr2f5el611/el611 (TU) standard conditions Prim-25 pharyngeal arch 1 jag1b expression spatial pattern, ameliorated
Prim-25 pharyngeal arch 1 prrx1b expression spatial pattern, ameliorated
Prim-25 pharyngeal arch 2 jag1b expression increased distribution, abnormal
Prim-25 pharyngeal arch 2 prrx1b expression increased distribution, abnormal
Day 5 Meckel's cartilage morphology, ameliorated
Day 5 palatoquadrate arch increased size, abnormal
Day 5 quadrate-anguloarticular joint morphology, ameliorated
nr2f1bel523/+; nr2f2el506/el506; nr2f5el611/el611 (TU) standard conditions Prim-25 pharyngeal arch 1 hand2 expression increased distribution, abnormal
Prim-25 pharyngeal arch 2 hand2 expression increased distribution, abnormal
nr2f1bel523/el523; nr2f2el506/el506; nr2f5el611/el611 (TU) standard conditions Prim-25 maxilla dlx5a expression increased amount, abnormal
Prim-25 pharyngeal arch 1 dlx5a expression increased distribution, abnormal
Prim-25 pharyngeal arch 2 dlx5a expression increased distribution, abnormal
Day 5 palatoquadrate arch increased thickness, abnormal
nr2f2el506/el506; nr2f5el611/el611 (TU) standard conditions Prim-25 maxilla dlx6a expression increased amount, abnormal
Prim-25 pharyngeal arch 1 dlx6a expression increased distribution, abnormal
Prim-25 pharyngeal arch 2 dlx6a expression increased distribution, abnormal
Acknowledgments:
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Reprinted from Developmental Cell, 44(3), Barske, L., Rataud, P., Behizad, K., Del Rio, L., Cox, S.G., Crump, J.G., Essential Role of Nr2f Nuclear Receptors in Patterning the Vertebrate Upper Jaw, 337-347.e5, Copyright (2018) with permission from Elsevier. Full text @ Dev. Cell