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Fig. 2. Attenuating Disp1 activity specifically in Shh-producing cell phenocopies Disp1 hypomorphic mutants. (A-E) External facial morphology of E18.5 embryos. Disp1 conditional mutants (C,E) display a spectrum of midline facial defects that result in a pointed face and nose that are similar but slightly milder than Disp1^2/2, Shh^+/- and Disp1^C829F/2, Shh^+/- embryos (B,D). (F-J) Fgf8 in situ to demarcate the epithelium of the nasal pit. Two nasal pits, which are positioned well apart in wild type (F), are brought closer to the midline in Disp1^2/2, Shh^+/- and Disp1^2/2C, Shh^Cre/+ (G,H), and are fused at the midline in Disp1^2/C829F, Shh^+/- (I). The fusion of the nasal pits occurs at a more medial position in Disp1^C829F/2C, Shh^Cre/+ embryos (J). (K-O) Alcian Blue (non-mineralized cartilage) and Alizarin Red (mineralized cartilage and bone) stained skeletons of E18.5 embryos. The premaxilla and upper incisor are missing from all mutants. The premaxilla, upper incisor and parietal bone are missing from Disp1^2/C829F, Shh^+/- (N) but not in the conditional mutant Disp1^C829F/2C, Shh^Cre/+ (O). Midline facial defects are due to attenuation of Shh induction and signaling in the ventral forebrain. (P-Y) Whole-mount in situ of Ptch1 (P-T) and Shh (U-Y) at E9.5 show an absence of Ptch1 upregulation in the frontal nasal process of the mutants and a failure of Shh induction in the ventral forebrain.

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