Developmental disruptions underlying brain abnormalities in ciliopathies

Jiami Guo, Holden Higginbotham, Jingjun Li, Jackie Nichols, Josua Hirt, Vladimir Ghukasyan and E.S. Anton ()
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Jiami Guo: University of North Carolina School of Medicine
Holden Higginbotham: University of North Carolina School of Medicine
Jingjun Li: University of North Carolina School of Medicine
Jackie Nichols: University of North Carolina School of Medicine
Josua Hirt: University of North Carolina School of Medicine
Vladimir Ghukasyan: University of North Carolina School of Medicine
E.S. Anton: University of North Carolina School of Medicine

Nature Communications, 2015, vol. 6, issue 1, 1-13

Abstract: Abstract Primary cilia are essential conveyors of signals underlying major cell functions. Cerebral cortical progenitors and neurons have a primary cilium. The significance of cilia function for brain development and function is evident in the plethora of developmental brain disorders associated with human ciliopathies. Nevertheless, the role of primary cilia function in corticogenesis remains largely unknown. Here we delineate the functions of primary cilia in the construction of cerebral cortex and their relevance to ciliopathies, using an shRNA library targeting ciliopathy genes known to cause brain disorders, but whose roles in brain development are unclear. We used the library to query how ciliopathy genes affect distinct stages of mouse cortical development, in particular neural progenitor development, neuronal migration, neuronal differentiation and early neuronal connectivity. Our results define the developmental functions of ciliopathy genes and delineate disrupted developmental events that are integrally related to the emergence of brain abnormalities in ciliopathies.

Date: 2015
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DOI: 10.1038/ncomms8857

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