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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{Delta}2/{Delta}2 (B), are rescued in Disp1{Delta}2/{Delta}2, Ptch1+/– (D), but exacerbated in Disp1{Delta}2/C829F (C), and Disp1{Delta}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{Delta}2/{Delta}2 (G), but restored in Disp1{Delta}2/{Delta}2, Ptch1+/– (I). The premaxilla, upper incisor and parietal bones are missing from Disp1{Delta}2/C829F and Disp1{Delta}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{Delta}2/{Delta}2 (L), fused in the midline in Disp1{Delta}2/C829F (M), and Disp1{Delta}2/C829F, Shh+/– (O), and are separated to normal distance in Disp1{Delta}2/{Delta}2, Ptch1+/– (N).





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