caa a22 4500 606175245 CHVBK 20210128100742.0 cr unu---uuuuu 210128e20150501xx s 000 0 eng 10.1007/s00018-014-1766-1 doi (NATIONALLICENCE)springer-10.1007/s00018-014-1766-1 Pax6 controls centriole maturation in cortical progenitors through Odf2 [Elektronische Daten] [Marco Tylkowski, Kefei Yang, Sigrid Hoyer-Fender, Anastassia Stoykova] Cortical glutamatergic neurons are generated by radial glial cells (RGCs), specified by the expression of transcription factor (TF) Pax6, in the germinative zones of the dorsal telencephalon. Here, we demonstrate that Pax6 regulates the structural assembly of the interphase centrosomes. In the cortex of the Pax6-deficient Small eye (Sey/Sey) mutant, we find a defect of the appendages of the mother centrioles, indicating incomplete centrosome maturation. Consequently, RGCs fail to generate primary cilia, and instead of staying in the germinative zone for renewal, RGCs detach from the ventricular surface thus affecting the interkinetic nuclear migration and they exit prematurely from mitosis. Mechanistically, we show that TF Pax6 directly regulates the activity of the Odf2 gene encoding for the appendage-specific protein Odf2 with a role for the assembly of mother centriole. Our findings demonstrate a molecular mechanism that explains important characteristics of the centrosome disassembly and malfunctioning in developing cortex lacking Pax6. Springer Basel, 2014 Centriole structure nationallicence Transcriptional control nationallicence Tylkowski Marco Research Group of Molecular Developmental Neurobiology, Department Molecular Cell Biology, Max-Planck Institute for Biophysical Chemistry, Am Faßberg 11, 37077, Göttingen, Germany aut Yang Kefei Johann-Friedrich-Blumenbach-Institute of Zoology and Anthropology, Developmental Biology, GZMB, Ernst-Caspari-Haus, Georg-August-Universität Göttingen, Justus-von-Liebig-Weg 11, Göttingen, Germany aut Hoyer-Fender Sigrid Johann-Friedrich-Blumenbach-Institute of Zoology and Anthropology, Developmental Biology, GZMB, Ernst-Caspari-Haus, Georg-August-Universität Göttingen, Justus-von-Liebig-Weg 11, Göttingen, Germany aut Stoykova Anastassia Research Group of Molecular Developmental Neurobiology, Department Molecular Cell Biology, Max-Planck Institute for Biophysical Chemistry, Am Faßberg 11, 37077, Göttingen, Germany aut Cellular and Molecular Life Sciences Springer Basel 72/9(2015-05-01), 1795-1809 1420-682X 72:9<1795 2015 72 18 https://doi.org/10.1007/s00018-014-1766-1 text/html Onlinezugriff via DOI BK010053 XK010053 XK010000 Metadata rights reserved Springer special CC-BY-NC licence nationallicence 1 research-article jats NATIONALLICENCE NATIONALLICENCE NL-springer NATIONALLICENCE 856 40 https://doi.org/10.1007/s00018-014-1766-1 text/html Onlinezugriff via DOI NATIONALLICENCE 700 1- Tylkowski Marco Research Group of Molecular Developmental Neurobiology, Department Molecular Cell Biology, Max-Planck Institute for Biophysical Chemistry, Am Faßberg 11, 37077, Göttingen, Germany aut NATIONALLICENCE 700 1- Yang Kefei Johann-Friedrich-Blumenbach-Institute of Zoology and Anthropology, Developmental Biology, GZMB, Ernst-Caspari-Haus, Georg-August-Universität Göttingen, Justus-von-Liebig-Weg 11, Göttingen, Germany aut NATIONALLICENCE 700 1- Hoyer-Fender Sigrid Johann-Friedrich-Blumenbach-Institute of Zoology and Anthropology, Developmental Biology, GZMB, Ernst-Caspari-Haus, Georg-August-Universität Göttingen, Justus-von-Liebig-Weg 11, Göttingen, Germany aut NATIONALLICENCE 700 1- Stoykova Anastassia Research Group of Molecular Developmental Neurobiology, Department Molecular Cell Biology, Max-Planck Institute for Biophysical Chemistry, Am Faßberg 11, 37077, Göttingen, Germany aut NATIONALLICENCE 773 0- Cellular and Molecular Life Sciences Springer Basel 72/9(2015-05-01), 1795-1809 1420-682X 72:9<1795 2015 72 18