Fig. 1. The Disp1 gene genetically interacts with Hh-signaling pathway
components, including Ptch1 and Shh gene. External facial
morphology of E18.5 embryos (A-E). The midline facial defects, clearly visible
in Disp1
2/
2 (B), are
rescued in Disp1
2/
2,
Ptch1+/ (D), but exacerbated in
Disp1
2/C829F (C), and
Disp1
2/C829F, Shh+/ (E).
Alcian Blue- and Alizarin Red-stained skeletons of E18.5 embryos (F-J). The
premaxilla and upper incisor are missing from
Disp1
2/
2 (G), but restored
in Disp1
2/
2,
Ptch1+/ (I). The premaxilla, upper incisor and parietal bones
are missing from Disp1
2/C829F and
Disp1
2/C829F, Shh+/ (H,J).
Fgf8 in situ to demarcate the epithelium of the nasal pit (K-O). Two
nasal pits, which are positioned well apart in wild type (K), are brought
closer to the midline in
Disp1
2/
2 (L), fused in the
midline in Disp1
2/C829F (M), and
Disp1
2/C829F, Shh+/ (O),
and are separated to normal distance in
Disp1
2/
2,
Ptch1+/ (N).