Xenbase Image ID: 151249

Figure 2. Knockdown of DS-epi1 reduces dermatan sulfate epimerase activity and neural crest-derived structures. (A) Morpholino oligonucleotides target the translation initiation sites of Dse and Dsel. (B) Endogenous DS epimerase activity is substantially decreased by Dse-MO and only little by Dsel-MO in stage 25 embryos. (C) Epimerase activity induced by the injection of 1 ng Dse mRNA is blocked by Dse-MO, but not by control-MO and Dsel-MO. The activity of 1 ng non-targeted Dse* mRNA is not affected by Dse-MO. (D) Tadpole at stage 40 injected with control-MO. (E,F) Microinjection of Dse-MO, but not Dse-5MM-MO, induces small eyes, a lack of dorsal fin (arrowheads), and reduced melanocyte formation (arrow). (G,H) Transversal trunk sections of stage 38 embryos following hematoxylin-eosin staining. Note the lack of a dorsal fin (arrowhead), dorsally approaching somites and hypoplastic notochord in the Dse-morphant embryo. (I-K) Ventral view of head skeletons at stage 45 in a schematic overview (I) and following alcian blue staining (J,K). Injection of Dse-MO, but not control-MO, causes a reduction of NC-derived cartilage structures. br, branchial segment; hy, hyoid segment; df, dorsal fin; ma, mandibular segment; no, notochord; nt, neural tube; so, somite. Indicated phenotypes are shown as follows: D, 70/70; E, 71/114 (microcephaly), 92/114 (reduced dorsal fin), 70/114 (less melanocytes); F, 63/63; G, 4/4; H, 4/4; J, 25/25; and K, 20/20. Image published in: Gouignard N et al. (2016) © 2016. 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 + dse MO + NF12 (Other Detection Assay) Fig.2 B Anatomical Phenotype abnormal molecular function null Disease Ehlers-Danlos syndrome Xla Wt + dse MO + NF25 (Other Detection Assay) Fig.2 B Anatomical Phenotype abnormal molecular function null Disease Ehlers-Danlos syndrome Xla Wt + dse MO + NF40 (whole-mount microscopy) Fig.2 E Anatomical Phenotype abnormal bending of tail abnormally decreased number of melanophore absent dorsal fin decreased length of tail decreased size of the eye decreased size of the head Disease Ehlers-Danlos syndrome Xla Wt + dse MO + NF40 (histology) Fig.2 H Anatomical Phenotype absent dorsal fin decreased size of the notochord Disease Ehlers-Danlos syndrome Xla Wt + dse MO + NF40 (histology) Fig.2 I, K. Anatomical Phenotype abnormally decreased mass of chondrocranium abnormally decreased volume of chondrocranium decreased size of the branchial arch skeleton decreased size of the chondrocranium decreased size of the hyoid arch skeleton decreased size of the mandibular arch Disease Ehlers-Danlos syndrome Larger Image Printer Friendly View

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