Arl13b in Primary Cilia Regulates the Migration and Placement of Interneurons in the Developing Cerebral Cortex
| Autor: | Holden Higginbotham, Tae Yeon Eom, Tamara Caspary, Amelia Bachleda, Vladimir Gukassyan, Laura E. Mariani, Corey L. Cusack, Joshua Hirt, Eva S. Anton, Cary Lai |
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| Rok vydání: | 2012 |
| Předmět: |
genetic structures
Interneuron Mice Transgenic Biology Article Retina General Biochemistry Genetics and Molecular Biology Joubert syndrome Interneuron migration Mice 03 medical and health sciences 0302 clinical medicine Cell Movement Cerebellar Diseases Interneurons Live cell imaging Cerebellum medicine Animals Humans Abnormalities Multiple Cilia Eye Abnormalities Receptor Molecular Biology Process (anatomy) 030304 developmental biology Cerebral Cortex 0303 health sciences ADP-Ribosylation Factors musculoskeletal neural and ocular physiology Cilium fungi Cell Biology Anatomy Kidney Diseases Cystic medicine.disease medicine.anatomical_structure nervous system Cerebral cortex Mutant Proteins Neuroscience 030217 neurology & neurosurgery Developmental Biology |
| Zdroj: | Developmental Cell. 23(5):925-938 |
| ISSN: | 1534-5807 |
| DOI: | 10.1016/j.devcel.2012.09.019 |
| Popis: | SummaryCoordinated migration and placement of interneurons and projection neurons lead to functional connectivity in the cerebral cortex; defective neuronal migration and the resultant connectivity changes underlie the cognitive defects in a spectrum of neurological disorders. Here we show that primary cilia play a guiding role in the migration and placement of postmitotic interneurons in the developing cerebral cortex and that this process requires the ciliary protein, Arl13b. Through live imaging of interneuronal cilia, we show that migrating interneurons display highly dynamic primary cilia and we correlate cilia dynamics with the interneuron's migratory state. We demonstrate that the guidance cue receptors essential for interneuronal migration localize to interneuronal primary cilia, but their concentration and dynamics are altered in the absence of Arl13b. Expression of Arl13b variants known to cause Joubert syndrome induce defective interneuronal migration, suggesting that defects in cilia-dependent interneuron migration may in part underlie the neurological defects in Joubert syndrome patients.Video Abstract |
| Databáze: | OpenAIRE |
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