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Updated: May 16, 2026
07:07
Evaluation of Planar-Cell-Polarity Phenotypes in Ciliopathy Mouse Mutant Cochlea
Published on: February 21, 2016
Wtip and Vangl2 are required for mitotic spindle orientation and cloaca morphogenesis
Ekaterina Bubenshchikova1, Koichiro Ichimura, Yayoi Fukuyo
1Department of Cell Biology, University of Oklahoma Health Science Center , Oklahoma City, OK 73104 , USA ; Department of Medicine and Rammelkamp Center for Education and Research, MetroHealth Medical Center, Case Western Reserve University School of Medicine , Cleveland, OH 44109 , USA.
Biology Open
|December 6, 2012
Summary
Wtip protein is essential for proper cilia formation and cell division during embryonic development. Loss of Wtip causes kidney cysts and other developmental defects, suggesting a role in ciliopathies.
Area of Science:
- Developmental Biology
- Cell Biology
- Genetics
Background:
- Cilia and basal body dysfunction cause ciliopathies, leading to kidney cyst formation.
- Cell division defects are observed in cystic kidneys, but mechanisms are unclear.
- The role of Wtip in ciliogenesis during embryonic development is unknown.
Purpose of the Study:
- To investigate the role of Wtip in ciliogenesis and embryonic development.
- To determine the function of Wtip in basal body and cilia formation.
- To elucidate the molecular mechanisms underlying Wtip-associated developmental defects.
Main Methods:
- Zebrafish model system.
- Knockdown of Wtip expression using morpholino.
- Phenotypic analysis of embryonic development.
- Assessment of mitotic spindle orientation and cilia structure.
Main Results:
- Wtip knockdown causes pronephric cyst formation, hydrocephalus, and other developmental abnormalities.
- Wtip is enriched in basal bodies.
- Knockdown embryos exhibit defects in mitotic spindle orientation, ciliated cell number, and cilia motility.
- Wtip loss-of-function phenocopies Vangl2 (a planar cell polarity protein) loss-of-function, and they interact genetically.
Conclusions:
- Wtip is crucial for mitotic spindle orientation, cloaca morphogenesis, and planar cell polarity (PCP) in zebrafish.
- Wtip's function is essential for preventing developmental defects associated with ciliopathies.
- Wtip may be a key factor in the molecular etiology of certain ciliopathies.
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