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FIGURE 3. WHS related gene depletion affects craniofacial morphology. (A–E) Frontal views of 3dpf embryos (st. 40) following WHS gene single KD. (F–I) Measurements for facial width, height, midface area, and midface angle. A significant 6.54% increase in facial width and 11.43% increase in midface area were observed for Whsc1 KD. Whsc2 KD caused a 12.01% reduction in facial width and a 6.79% reduction in midface area. Letm1 KD caused a 10.33% decrease in facial width and a 8.49% decrease in midface area. Tacc3 KD caused a 21.27% decrease in facial width and a 16.33% decrease in midface area, and an 8.27% decrease in midface angle. Significance determined using a student’s unpaired t-test. (Embryos quantified: Control = 137, Whsc1 KD = 100, Whsc2 KD = 185, Letm1 KD = 115, Tacc3 KD = 79.) ∗∗∗∗P < 0.0001, ∗∗∗P < 0.001, ∗∗P < 0.01, n.s., not significant. Scalebar = 250 μm. Image published in: Mills A et al. (2019) Copyright © 2019 Mills, Bearce, Cella, Kim, Selig, Lee and Lowery. This image is reproduced with permission of the journal and the copyright holder. This is an open-access article distributed under the terms of the Creative Commons Attribution license Experiment + Assay Source Phenotypes and Disease Xla Wt + nsd2 MO + NF40 (whole-mount microscopy) Fig. 3B, FGHI Anatomical Phenotype abnormal craniofacial region morphology increased size of the head Disease Wolf-Hirschhorn syndrome Xla Wt + nelfa MO + NF40 (whole-mount microscopy) Fig. 3C, FGHI Anatomical Phenotype abnormal craniofacial region morphology decreased size of the head Disease Wolf-Hirschhorn syndrome Xla Wt + letm1 MO + NF40 (whole-mount microscopy) Fig. 3D, FGHI Anatomical Phenotype abnormal craniofacial region morphology decreased size of the head Disease Wolf-Hirschhorn syndrome Xla Wt + tacc3 MO + NF40 (whole-mount microscopy) Fig. 3E, FGHI Anatomical Phenotype abnormal craniofacial region morphology decreased size of the head Disease Wolf-Hirschhorn syndrome Larger Image Printer Friendly View

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