Morphogenetic defects underlie Superior Coloboma, a newly identified closure disorder of the dorsal eye
Fig 9
Aberrant SOS closure leads to abnormal vasculature.
(A) Surface projections of 26 hpf Tg(rx3:GFP; kdrl:mCherry) wildtype and gdf6a-/- embryos, shown without vessels (top row) and with vessels (bottom row). Last column shows expanded views of same gdf6a-/- eye, highlighting the divot in the dorsal retina at the inferior edge of the superior ocular sulcus (yellow arrow). Small panel is 90° lateral rotation of vessel in adjacent panel, showing the DRV turn and extend toward the hyaloid vasculature. (B) Surface projections of Tg(rx3:GFP; kdrl:mCherry) embryos before (22 hpf) and after (26 hpf) DRV formation, with and without cyclopamine treatment. (C-D) Quantification of the area and number of DRV vessel(s) in control and cyclopamine-treated 26 hpf embryos. n = number of embryos. Scale bars are 50 μm unless otherwise noted. Di-Pr, distal-proximal.