Fig. 1

Hh pathway mutations sensitize embryos to ethanol‐ or PBO‐induced craniofacial defects. (A–D) Flat‐mount preparations of 5 dpf neurocrania showing the range of phenotypes observed. Phenotypes ranged from apparently normal to severe, with increasing severity determined by more extensive malformations of the bilateral trabeculae. All images were captured at 10X magnification. (A′–D′) 20X magnification of the right trabeculae of A–D shows cell arrangement defects in affected embryos, specifically the stacking defect in mild embryos (B′) compared to normal stacking (A′). (E–F) Wild‐type or embryos with a single‐allele mutation in shha (tq252) were exposed to 0% 0.75% or 1% ethanol (E) or 0, 6.25, or 12.5 μM PBO (F). Percent malformations (mild, moderate, and severe) are shown. Incidence of malformations was compared between genotypes for each treatment group using Fisher’s exact test with Bonferroni correction for multiple comparisons. N ≥ 15 embryos per genotype per treatment.