title: Ptbp1 and Exosc9 knockdowns trigger skin stability defects through different pathways.
abstract: In humans, genetic diseases affecting skin integrity (genodermatoses) are generally caused by mutations in a small number of genes that encode structural components of the dermal-epidermal junctions. In this article, we first show that inactivation of both exosc9, which encodes a component of the RNA exosome, and ptbp1, which encodes an RNA-binding protein abundant in Xenopus embryonic skin, impairs embryonic Xenopus skin development, with the appearance of dorsal blisters along the anterior part of the fin. However, histological and electron microscopy analyses revealed that the two phenotypes are distinct. Exosc9 morphants are characterized by an increase in the apical surface of the goblet cells, loss of adhesion between the sensorial and peridermal layers, and a decrease in the number of ciliated cells within the blisters. Ptbp1 morphants are characterized by an altered goblet cell morphology. Gene expression profiling by deep RNA sequencing showed that the expression of epidermal and genodermatosis-related genes is also differentially affected in the two morphants, indicating that alterations in post-transcriptional regulations can lead to skin developmental defects through different routes. Therefore, the developing larval epidermis of Xenopus will prove to be a useful model for dissecting the post-transcriptional regulatory network involved in skin development and stability with significant implications for human diseases.
authors.affiliation: CNRS, UMR 6290 Institut Génétique et Développement de Rennes, Université de Rennes 1, Rennes, France.
authors.forename: Maud
authors.initials: M
authors.lastname: Noiret
authors.affiliation: CNRS, UMR 6290 Institut Génétique et Développement de Rennes, Université de Rennes 1, Rennes, France.
authors.forename: Stéphanie
authors.initials: S
authors.lastname: Mottier
authors.affiliation: CNRS, UMR 6290 Institut Génétique et Développement de Rennes, Université de Rennes 1, Rennes, France.
authors.forename: Gaelle
authors.initials: G
authors.lastname: Angrand
authors.affiliation: CNRS, UMR 6290 Institut Génétique et Développement de Rennes, Université de Rennes 1, Rennes, France.
authors.forename: Carole
authors.initials: C
authors.lastname: Gautier-Courteille
authors.affiliation: CNRS, UMR 6290 Institut Génétique et Développement de Rennes, Université de Rennes 1, Rennes, France.
authors.forename: Hubert
authors.initials: H
authors.lastname: Lerivray
authors.affiliation: CNRS, UMR 6290 Institut Génétique et Développement de Rennes, Université de Rennes 1, Rennes, France.
authors.forename: Justine
authors.initials: J
authors.lastname: Viet
authors.affiliation: CNRS, UMR 6290 Institut Génétique et Développement de Rennes, Université de Rennes 1, Rennes, France.
authors.forename: Luc
authors.initials: L
authors.lastname: Paillard
authors.affiliation: CNRS, UMR 6290 Institut Génétique et Développement de Rennes, Université de Rennes 1, Rennes, France.
authors.forename: Agnes
authors.initials: A
authors.lastname: Mereau
authors.affiliation: CNRS, UMR 6290 Institut Génétique et Développement de Rennes, Université de Rennes 1, Rennes, France.
authors.forename: Serge
authors.initials: S
authors.lastname: Hardy
authors.affiliation: CNRS, UMR 6290 Institut Génétique et Développement de Rennes, Université de Rennes 1, Rennes, France. Electronic address: yann.audic@univ-rennes1.fr.
authors.forename: Yann
authors.initials: Y
authors.lastname: Audic
mesh_terms: D058500:Animal Fins / Q000196:embryology; D000818:Animals; D004625:Embryo, Nonmammalian / Q000187:drug effects / Q000473:pathology / Q000648:ultrastructure; D004817:Epidermis / Q000187:drug effects / Q000473:pathology / Q000648:ultrastructure; D063326:Exosome Multienzyme Ribonuclease Complex / Q000235:genetics* / Q000378:metabolism; D020869:Gene Expression Profiling; D018507:Gene Expression Regulation, Developmental / Q000187:drug effects; D055785:Gene Knockdown Techniques; D053263:Gene Regulatory Networks / Q000187:drug effects; D034441:Heterogeneous-Nuclear Ribonucleoproteins / Q000235:genetics* / Q000378:metabolism; D017403:In Situ Hybridization; D060172:Morpholinos / Q000494:pharmacology; D038941:Polypyrimidine Tract-Binding Protein / Q000235:genetics* / Q000378:metabolism; D016601:RNA-Binding Proteins / Q000235:genetics* / Q000378:metabolism; D015398:Signal Transduction / Q000187:drug effects / Q000235:genetics; D012867:Skin / Q000196:embryology* / Q000473:pathology*; D029867:Xenopus Proteins / Q000235:genetics* / Q000378:metabolism; D014982:Xenopus laevis / Q000196:embryology*
chemical_list: D034441:Heterogeneous-Nuclear Ribonucleoproteins; D060172:Morpholinos; D016601:RNA-Binding Proteins; D029867:Xenopus Proteins; C000608280:polypyrimidine tract binding protein 1, Xenopus; D038941:Polypyrimidine Tract-Binding Protein; C000608281:Exosc9 protein, Xenopus; D063326:Exosome Multienzyme Ribonuclease Complex
keywords: Epidermis; Genodermatosis; Post-transcriptional regulations; RNA-binding proteins