Wtip and Vangl2 are required for mitotic spindle orientation and cloaca morphogenesis

Author:

Bubenshchikova Ekaterina12,Ichimura Koichiro13,Fukuyo Yayoi1,Powell Rebecca1,Hsu Chaonan1,Morrical Stephen O.2,Sedor John R.2,Sakai Tatsuo3,Obara Tomoko12

Affiliation:

1. Department of Cell Biology, University of Oklahoma Health Science Center, Oklahoma City, OK 73104, USA

2. Department of Medicine and Rammelkamp Center for Education and Research, MetroHealth Medical Center, Case Western Reserve University School of Medicine, Cleveland, OH 44109, USA

3. Department of Anatomy and Life Structure, Juntendo University School of Medicine, Tokyo 113-8421, Japan

Abstract

Summary Defects in cilia and basal bodies function are linked to ciliopathies, which result in kidney cyst formation. Recently, cell division defects have been observed in cystic kidneys, but the underlying mechanisms of such defects remain unclear. Wtip is an LIM domain protein of the Ajuba/Zyxin family, but its role in ciliogenesis during embryonic development has not been previously described. We report Wtip is enriched in the basal body and knockdown of wtip leads to pronephric cyst formation, cloaca malformation, hydrocephalus, body curvature, and pericardial edema. We additionally show that wtip knockdown embryos display segment-specific defects in the pronephros: mitotic spindle orientation defects are observed only in the anterior and middle pronephros; cloaca malformation is accompanied by a reduced number of ciliated cells; and ciliated cells lack the striated rootlet that originates from basal bodies, which results in a lack of cilia motility. Our data suggest that loss of Wtip function phenocopies Vangl2 loss of function, a core planar cell polarity (PCP) protein located in the basal body protein. Furthermore, we demonstrate that wtip and vangl2 interact genetically. Taken together, our results indicate that in zebrafish, Wtip is required for mitotic spindle orientation in the anterior and middle of the pronephros, cloaca morphogenesis, and PCP, which may underlie the molecular etiology of ciliopathies.

Publisher

The Company of Biologists

Subject

General Agricultural and Biological Sciences,General Biochemistry, Genetics and Molecular Biology

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