title: FMR1/FXR1 and the miRNA pathway are required for eye and neural crest development.
abstract: FMR1 and FXR1 are RNA binding proteins interacting with the miRNA-induced silencing complex, RISC. Here we describe for the first time the function of these proteins during eye and neural crest (NC) development in Xenopus laevis. A loss of FMR1 or FXR1 results in abnormal eye development as well as defects in cranial cartilage derived from cranial NC cells. We further investigated the possible mechanism of these phenotypes by showing that a depletion of Dicer, an important enzyme for generating all mature miRNAs, in the anterior neural tissue also leads to eye and cranial cartilage defects. Furthermore, we examined the function of 12 miRNAs during anterior neural development. We show a specific requirement of six selected miRNAs during eye and cranial cartilage development. Mir-130a, -219, and -23b are involved in eye formation only whereas loss of miR-200b, miR-96 and miR-196a results in strong defects during eye as well as cranial cartilage development. Our results suggest an essential role for FMR1 and FXR1 for eye and NC development in X.laevis likely through an interaction with the miRNA pathway.
authors.affiliation: Institute for Biochemistry and Molecular Biology, Ulm University, Albert-Einstein-Allee 11, D-89081 Ulm, Germany.
authors.forename: Susanne
authors.initials: S
authors.lastname: Gessert
authors.forename: Verena
authors.initials: V
authors.lastname: Bugner
authors.forename: Aleksandra
authors.initials: A
authors.lastname: Tecza
authors.forename: Maximilian
authors.initials: M
authors.lastname: Pinker
authors.forename: Michael
authors.initials: M
authors.lastname: Kühl
mesh_terms: D000818:Animals; D015536:Down-Regulation; D005123:Eye / Q000196:embryology*; D051860:Fragile X Messenger Ribonucleoprotein 1 / Q000378:metabolism*; D055785:Gene Knockdown Techniques; D035683:MicroRNAs / Q000378:metabolism*; D009432:Neural Crest / Q000196:embryology*; D055495:Neurogenesis; D016601:RNA-Binding Proteins / Q000378:metabolism*; D043244:Ribonuclease III / Q000378:metabolism; D029867:Xenopus Proteins / Q000378:metabolism*; D014982:Xenopus laevis / Q000196:embryology*
chemical_list: D051860:Fragile X Messenger Ribonucleoprotein 1; D035683:MicroRNAs; D016601:RNA-Binding Proteins; D043244:Ribonuclease III; D029867:Xenopus Proteins; C505711:FXR1 protein, Xenopus; C497686:Fmr1 protein, Xenopus