Morphogenetic defects underlie Superior Coloboma, a newly identified closure disorder of the dorsal eye
Fig 5
Inhibition of Hedgehog signaling rescues closure of the superior ocular sulcus in Gdf6a-deficient embryos.
(A-B) Effect of Hedgehog inhibition (cyclopamine treatment) on SOS closure in Gdf6a-deficient embryos. DIC images of gdf6a+/- eyes, treated with either control solution (left) or 10 μM cyclopamine (right) (A). SOS marked by red asterisk. Quantification of effect of cyclopamine treatment on SOS closure in gdf6a+/- incross embryos (B). (C-D) Effect of cyclopamine on dorsal retinal patterning in Gdf6a-deficient embryos. tbx5 RNA expression in eyes from 28 hpf gdf6a+/+, gdf6a+/-, and gdf6a-/- embryos with or without cyclopamine treatment (C). Quantification of effect of cyclopamine treatment on area of tbx5 expression (D). n = number of embryos, N = 4 (B) or 3 (D) experiments. Data are means ± SEM. Data in B and D are means ± SEM; Statistics in B is a one-way ANOVA with Tukey’s test, D is two-way ANOVA with Tukey’s test: **P<0.01. Scale bars are 50 μm.