Durán Alonso, BeatrizVendrell, VíctorLópez Hernández, IrisAlonso Pérez, María TeresaMartin, Donna M.Giráldez, FernandoCarramolino, LauraGiovinazzo, GiovannaVázquez, EnriqueTorres, Miguel S.Schimmang, Thomas2021-07-212021-07-212021Durán Alonso MB, Vendrell V, López-Hernández I, Alonso MT, Martin DM, Giráldez F, Carramolino L, Giovinazzo G, Vázquez E, Torres M, Schimmang T. Meis2 is required for inner ear formation and proper morphogenesis of the cochlea. Front Cell Dev Biol. 2021;9:679325. DOI: 10.3389/fcell.2021.6793252296-634Xhttp://hdl.handle.net/10230/48259Meis genes have been shown to control essential processes during development of the central and peripheral nervous system. Here we have explored the roles of the Meis2 gene during vertebrate inner ear induction and the formation of the cochlea. Meis2 is expressed in several tissues required for inner ear induction and in non-sensory tissue of the cochlear duct. Global inactivation of Meis2 in the mouse leads to a severely reduced size of the otic vesicle. Tissue-specific knock outs of Meis2 reveal that its expression in the hindbrain is essential for otic vesicle formation. Inactivation of Meis2 in the inner ear itself leads to an aberrant coiling of the cochlear duct. By analyzing transcriptomes obtained from Meis2 mutants and ChIPseq analysis of an otic cell line, we define candidate target genes for Meis2 which may be directly or indirectly involved in cochlear morphogenesis. Taken together, these data show that Meis2 is essential for inner ear formation and provide an entry point to unveil the network underlying proper coiling of the cochlear duct.application/pdfeng© 2021 Durán Alonso, Vendrell, López-Hernández, Alonso, Martin, Giráldez, Carramolino, Giovinazzo, Vázquez, Torres and Schimmang. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.info:eu-repo/semantics/articlehttp://dx.doi.org/10.3389/fcell.2021.679325MeisCochleaInner earMouseOrgan of cortiinfo:eu-repo/semantics/openAccess