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Fig. 1. ATP4 is required for normal development of the embryonic epidermis. (A and B) Morphological analysis of the embryonic epidermis at stage 32 in control morpholino oligonucleotide (CoMO) (A) and atp4aMO (B) injected specimens. Knockdown of atp4a lead to defects in ciliation of multiciliated cells (MCCs, green) and reduced numbers of small secretory cells (SSCs, red), but without obvious effects on ion secreting cells (ISCs, yellow) or outer/goblet cells (blue). (C) Knockdown of atp4a attenuated foxj1 and foxa1 expression, but not foxi1 expression in the skin epidermis. Embryos were unilaterally injected with atp4aMO at the four-cell stage and assayed for foxj1/foxa1/foxi1 expression by WMISH at stage 15. Correct targeting was confirmed by co-injection of lineage tracer. Depicted embryos are derived from the same injected batch. Numbers in the right lower corner indicate frequency of phenotype (a, anterior; d, dorsal; p, posterior; st., stage; v, ventral). Image published in: Walentek P et al. (2015) Copyright © 2015. Image reproduced with permission of the Publisher, Elsevier B. V. Gene Synonyms Species Stage(s) Tissue foxa1.L fkh2, foxa1-a, foxa1-b, foxa1b, HNF-3 alpha, HNF3 alpha, HNF3-alpha, hnf3a, HNF3alpha, tcf3a, tFoxA1, xfkh2 X. laevis Throughout NF stage 15 epidermis secretory epithelial cell foxj1.L foxj1-a, foxj1-b, hfh-4, hfh4, xfoxj1 X. laevis Throughout NF stage 15 epidermis ciliated epidermal cell multiciliated cell foxi1.L ectodermally-expressed mesendoderm antagonist, ema, FoxI1e, foxi1e, foxi3, Xema, xfoxi1 X. laevis Throughout NF stage 15 epidermis ionocyte Image source: Published Experiment + Assay Source Phenotypes and Disease Xla Wt + atp4a MO + NF32 (scanning electron microscopy) Fig. 1 B Anatomical Phenotype abnormally decreased number of cilium in the multiciliated cell abnormally decreased number of small secretory cell decreased size of the cilium in the multiciliated epidermal cell Disease pneumonia Xla Wt + atp4a MO + NF15-18 (in situ hybridization) Fig. 1 C row 1 col 2 Disease pneumonia Expression Phenotype decreased amount foxj1.L expression in epidermis Xla Wt + atp4a MO + NF15-18 (in situ hybridization) Fig. 1 C row 2 col 2 Disease pneumonia Expression Phenotype decreased amount foxa1.L expression in epidermis Larger Image Printer Friendly View

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