دورية أكاديمية
WD40-repeat 47, a microtubule-associated protein, is essential for brain development and autophagy.
| العنوان: | WD40-repeat 47, a microtubule-associated protein, is essential for brain development and autophagy. |
|---|---|
| المؤلفون: | Kannan M; Department of Translational Medicine and Neurogenetics, Institut de Génétique et de Biologie Moléculaire et Cellulaire, 67404 Illkirch, France.; Centre National de la Recherche Scientifique, UMR7104, 67404 Illkirch, France.; Institut National de la Santé et de la Recherche Médicale, U964, 67404 Illkirch, France.; Université de Strasbourg, 67404 Illkirch, France.; Center for Integrative Genomics, University of Lausanne, CH-1015 Lausanne, Switzerland., Bayam E; Department of Translational Medicine and Neurogenetics, Institut de Génétique et de Biologie Moléculaire et Cellulaire, 67404 Illkirch, France.; Centre National de la Recherche Scientifique, UMR7104, 67404 Illkirch, France.; Institut National de la Santé et de la Recherche Médicale, U964, 67404 Illkirch, France.; Université de Strasbourg, 67404 Illkirch, France., Wagner C; Department of Translational Medicine and Neurogenetics, Institut de Génétique et de Biologie Moléculaire et Cellulaire, 67404 Illkirch, France.; Centre National de la Recherche Scientifique, UMR7104, 67404 Illkirch, France.; Institut National de la Santé et de la Recherche Médicale, U964, 67404 Illkirch, France.; Université de Strasbourg, 67404 Illkirch, France., Rinaldi B; Génétique Moléculaire Génomique Microbiologie, UMR7156, Université de Strasbourg, CNRS, 67000 Strasbourg, France., Kretz PF; Department of Translational Medicine and Neurogenetics, Institut de Génétique et de Biologie Moléculaire et Cellulaire, 67404 Illkirch, France.; Centre National de la Recherche Scientifique, UMR7104, 67404 Illkirch, France.; Institut National de la Santé et de la Recherche Médicale, U964, 67404 Illkirch, France.; Université de Strasbourg, 67404 Illkirch, France., Tilly P; Department of Translational Medicine and Neurogenetics, Institut de Génétique et de Biologie Moléculaire et Cellulaire, 67404 Illkirch, France.; Centre National de la Recherche Scientifique, UMR7104, 67404 Illkirch, France.; Institut National de la Santé et de la Recherche Médicale, U964, 67404 Illkirch, France.; Université de Strasbourg, 67404 Illkirch, France., Roos M; Department of Physiological Sciences, University of Stellenbosch, 7600 Stellenbosch, South Africa., McGillewie L; South African Medical Research Council Centre for Tuberculosis Research, Department of Biomedical Sciences, University of Stellenbosch, 7505 Tygerberg, South Africa., Bär S; Génétique Moléculaire Génomique Microbiologie, UMR7156, Université de Strasbourg, CNRS, 67000 Strasbourg, France., Minocha S; Center for Integrative Genomics, University of Lausanne, CH-1015 Lausanne, Switzerland., Chevalier C; Department of Translational Medicine and Neurogenetics, Institut de Génétique et de Biologie Moléculaire et Cellulaire, 67404 Illkirch, France.; Centre National de la Recherche Scientifique, UMR7104, 67404 Illkirch, France.; Institut National de la Santé et de la Recherche Médicale, U964, 67404 Illkirch, France.; Université de Strasbourg, 67404 Illkirch, France., Po C; ICube, UMR 7357, Fédération de Médecine Translationnelle, University of Strasbourg, 67085 Strasbourg, France., Chelly J; Department of Translational Medicine and Neurogenetics, Institut de Génétique et de Biologie Moléculaire et Cellulaire, 67404 Illkirch, France.; Centre National de la Recherche Scientifique, UMR7104, 67404 Illkirch, France.; Institut National de la Santé et de la Recherche Médicale, U964, 67404 Illkirch, France.; Université de Strasbourg, 67404 Illkirch, France., Mandel JL; Department of Translational Medicine and Neurogenetics, Institut de Génétique et de Biologie Moléculaire et Cellulaire, 67404 Illkirch, France.; Centre National de la Recherche Scientifique, UMR7104, 67404 Illkirch, France.; Institut National de la Santé et de la Recherche Médicale, U964, 67404 Illkirch, France.; Université de Strasbourg, 67404 Illkirch, France., Borgatti R; Neuropsychiatry and Neurorehabilitation Unit, Scientific Institute, Istituto di Ricovero e Cura a Carattere Scientifico Eugenio Medea, 23842 Bosisio Parini, Lecco, Italy., Piton A; Department of Translational Medicine and Neurogenetics, Institut de Génétique et de Biologie Moléculaire et Cellulaire, 67404 Illkirch, France.; Centre National de la Recherche Scientifique, UMR7104, 67404 Illkirch, France.; Institut National de la Santé et de la Recherche Médicale, U964, 67404 Illkirch, France.; Université de Strasbourg, 67404 Illkirch, France., Kinnear C; South African Medical Research Council Centre for Tuberculosis Research, Department of Biomedical Sciences, University of Stellenbosch, 7505 Tygerberg, South Africa., Loos B; Department of Physiological Sciences, University of Stellenbosch, 7600 Stellenbosch, South Africa., Adams DJ; Wellcome Trust Sanger Institute, Hinxton, CB10 1SA Cambridge, United Kingdom., Hérault Y; Department of Translational Medicine and Neurogenetics, Institut de Génétique et de Biologie Moléculaire et Cellulaire, 67404 Illkirch, France.; Centre National de la Recherche Scientifique, UMR7104, 67404 Illkirch, France.; Institut National de la Santé et de la Recherche Médicale, U964, 67404 Illkirch, France.; Université de Strasbourg, 67404 Illkirch, France., Collins SC; Department of Translational Medicine and Neurogenetics, Institut de Génétique et de Biologie Moléculaire et Cellulaire, 67404 Illkirch, France.; Centre National de la Recherche Scientifique, UMR7104, 67404 Illkirch, France.; Institut National de la Santé et de la Recherche Médicale, U964, 67404 Illkirch, France.; Université de Strasbourg, 67404 Illkirch, France.; Centre des Sciences du Goût et de l'Alimentation, Université de Bourgogne-Franche Comté, 21000 Dijon, France., Friant S; Génétique Moléculaire Génomique Microbiologie, UMR7156, Université de Strasbourg, CNRS, 67000 Strasbourg, France., Godin JD; Department of Translational Medicine and Neurogenetics, Institut de Génétique et de Biologie Moléculaire et Cellulaire, 67404 Illkirch, France.; Centre National de la Recherche Scientifique, UMR7104, 67404 Illkirch, France.; Institut National de la Santé et de la Recherche Médicale, U964, 67404 Illkirch, France.; Université de Strasbourg, 67404 Illkirch, France., Yalcin B; Department of Translational Medicine and Neurogenetics, Institut de Génétique et de Biologie Moléculaire et Cellulaire, 67404 Illkirch, France; Binnaz.Yalcin@igbmc.fr.; Centre National de la Recherche Scientifique, UMR7104, 67404 Illkirch, France.; Institut National de la Santé et de la Recherche Médicale, U964, 67404 Illkirch, France.; Université de Strasbourg, 67404 Illkirch, France. |
| مؤلفون مشاركون: | Sanger Mouse Genetics Project |
| المصدر: | Proceedings of the National Academy of Sciences of the United States of America [Proc Natl Acad Sci U S A] 2017 Oct 31; Vol. 114 (44), pp. E9308-E9317. Date of Electronic Publication: 2017 Oct 12. |
| نوع المنشور: | Journal Article; Research Support, Non-U.S. Gov't |
| اللغة: | English |
| بيانات الدورية: | Publisher: National Academy of Sciences Country of Publication: United States NLM ID: 7505876 Publication Model: Print-Electronic Cited Medium: Internet ISSN: 1091-6490 (Electronic) Linking ISSN: 00278424 NLM ISO Abbreviation: Proc Natl Acad Sci U S A Subsets: MEDLINE |
| أسماء مطبوعة: | Original Publication: Washington, DC : National Academy of Sciences |
| مواضيع طبية MeSH: | Autophagy/*physiology , Brain/*growth & development , Brain/*metabolism , Microfilament Proteins/*metabolism , Microtubule-Associated Proteins/*metabolism , WD40 Repeats/*physiology, Animals ; Cell Movement/physiology ; Cell Proliferation/physiology ; Cells, Cultured ; Female ; Male ; Mice ; Mice, Inbred C57BL ; Microtubules/metabolism ; Microtubules/physiology ; Neurogenesis/physiology ; Neurons/metabolism ; Neurons/physiology ; Phenotype ; Stem Cells/metabolism ; Stem Cells/physiology |
| مستخلص: | The family of WD40-repeat (WDR) proteins is one of the largest in eukaryotes, but little is known about their function in brain development. Among 26 WDR genes assessed, we found 7 displaying a major impact in neuronal morphology when inactivated in mice. Remarkably, all seven genes showed corpus callosum defects, including thicker ( Atg16l1 , Coro1c , Dmxl2 , and Herc1 ), thinner ( Kif21b and Wdr89 ), or absent corpus callosum ( Wdr47 ), revealing a common role for WDR genes in brain connectivity. We focused on the poorly studied WDR47 protein sharing structural homology with LIS1, which causes lissencephaly. In a dosage-dependent manner, mice lacking Wdr47 showed lethality, extensive fiber defects, microcephaly, thinner cortices, and sensory motor gating abnormalities. We showed that WDR47 shares functional characteristics with LIS1 and participates in key microtubule-mediated processes, including neural stem cell proliferation, radial migration, and growth cone dynamics. In absence of WDR47, the exhaustion of late cortical progenitors and the consequent decrease of neurogenesis together with the impaired survival of late-born neurons are likely yielding to the worsening of the microcephaly phenotype postnatally. Interestingly, the WDR47-specific C-terminal to LisH (CTLH) domain was associated with functions in autophagy described in mammals. Silencing WDR47 in hypothalamic GT1-7 neuronal cells and yeast models independently recapitulated these findings, showing conserved mechanisms. Finally, our data identified superior cervical ganglion-10 (SCG10) as an interacting partner of WDR47. Taken together, these results provide a starting point for studying the implications of WDR proteins in neuronal regulation of microtubules and autophagy. Competing Interests: The authors declare no conflict of interest. (Published under the PNAS license.) |
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| فهرسة مساهمة: | Investigator: VE Vancollie; LFE Anthony; SA Maguire; D Lafont; SA Pearson; AS Gates; M Sanderson; C Shannon; MT Sumowski; RSB McLaren-Jones; A Swiatkowska; CM Isherwood; EL Cambridge; HM Wilson; SS Caetano; AKB Maguire; A Galli; AO Speak; J Dench; E Tuck; J Estabel; A Green; C Tudor; E Siragher; M Dabrowska; CI Mazzeo; Y Hooks; F Kussy; M Griffiths; D Gannon; B Doe; K Boroviak; H Wardle-Jones; N Griggs; J Bottomley; E Ryder; D Gleeson; JK White; R Ramirez-Solis; CJ Lelliott Keywords: WD40-repeat proteins; autophagy; corpus callosum agenesis; microcephaly; neurogenesis |
| المشرفين على المادة: | 0 (Microfilament Proteins) 0 (Microtubule-Associated Proteins) |
| تواريخ الأحداث: | Date Created: 20171029 Date Completed: 20180618 Latest Revision: 20181113 |
| رمز التحديث: | 20231215 |
| مُعرف محوري في PubMed: | PMC5676932 |
| DOI: | 10.1073/pnas.1713625114 |
| PMID: | 29078390 |
| قاعدة البيانات: | MEDLINE |
| تدمد: | 1091-6490 |
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| DOI: | 10.1073/pnas.1713625114 |