Loss of Wdfy3 in mice alters cerebral cortical neurogenesis reflecting aspects of the autism pathology

Lori A. Orosco, Adam P. Ross, Staci L. Cates, Sean E. Scott, Dennis Wu, Jiho Sohn, David Pleasure, Samuel J. Pleasure, Iannis E. Adamopoulos and Konstantinos S Zarbalis ()
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Lori A. Orosco: University of California at Davis
Adam P. Ross: University of California at Davis
Staci L. Cates: University of California at Davis
Sean E. Scott: University of California at Davis
Dennis Wu: Institute for Pediatric Regenerative Medicine, Shriners Hospitals for Children, Northern California
Jiho Sohn: Institute for Pediatric Regenerative Medicine, Shriners Hospitals for Children, Northern California
David Pleasure: Institute for Pediatric Regenerative Medicine, Shriners Hospitals for Children, Northern California
Samuel J. Pleasure: Programs in Neuroscience, Developmental and Stem Cell Biology, UCSF Institute for Regeneration Medicine, University of California at San Francisco, Sandler Neurosciences Center
Iannis E. Adamopoulos: Institute for Pediatric Regenerative Medicine, Shriners Hospitals for Children, Northern California
Konstantinos S Zarbalis: University of California at Davis

Nature Communications, 2014, vol. 5, issue 1, 1-13

Abstract: Abstract Autism spectrum disorders (ASDs) are complex and heterogeneous developmental disabilities affecting an ever-increasing number of children worldwide. The diverse manifestations and complex, largely genetic aetiology of ASDs pose a major challenge to the identification of unifying neuropathological features. Here we describe the neurodevelopmental defects in mice that carry deleterious alleles of the Wdfy3 gene, recently recognized as causative in ASDs. Loss of Wdfy3 leads to a regionally enlarged cerebral cortex resembling early brain overgrowth described in many children on the autism spectrum. In addition, affected mouse mutants display migration defects of cortical projection neurons, a recognized cause of epilepsy, which is significantly comorbid with autism. Our analysis of affected mouse mutants defines an important role for Wdfy3 in regulating neural progenitor divisions and neural migration in the developing brain. Furthermore, Wdfy3 is essential for cerebral expansion and functional organization while its loss-of-function results in pathological changes characteristic of ASDs.

Date: 2014
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DOI: 10.1038/ncomms5692

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