Meghna Kannan, Efil Bayam, Christel Wagner, Bruno Rinaldi, Perrine F Kretz, Peggy Tilly, Marna Roos, Lara McGillewie, Séverine Bär, Shilpi Minocha, Claire Chevalier, Chrystelle Po, Jamel Chelly, Jean-Louis Mandel, Renato Borgatti, Amélie Piton, Craig Kinnear, Ben Loos, David J Adams, Yann Hérault, Stephan C Collins, Sylvie Friant, Juliette D Godin, Binnaz Yalcin
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...
October 31, 2017: Proceedings of the National Academy of Sciences of the United States of America