Intraflagellar Transport Proteins Are Essential for Cilia Formation and for Planar Cell Polarity

被引:51
|
作者
Cao, Ying [1 ]
Park, Alice [1 ]
Sun, Zhaoxia [1 ]
机构
[1] Yale Univ, Sch Med, Dept Genet, New Haven, CT 06520 USA
来源
JOURNAL OF THE AMERICAN SOCIETY OF NEPHROLOGY | 2010年 / 21卷 / 08期
关键词
POLYCYSTIC KIDNEY-DISEASE; KINESIN-II; ZEBRAFISH PRONEPHROS; CYSTIC KIDNEY; BETA-CATENIN; SENSORY NEURONS; TRANSGENIC MICE; REGULATES IFT; GASTRULATION; GENE;
D O I
10.1681/ASN.2009091001
中图分类号
R5 [内科学]; R69 [泌尿科学(泌尿生殖系疾病)];
学科分类号
1002 ; 100201 ;
摘要
The highly conserved intraflagellar transport (IFT) proteins are essential for cilia formation in multiple organisms, but surprisingly, cilia form in multiple zebrafish ift mutants. Here, we detected maternal deposition of ift gene products in zebrafish and found that ciliary assembly occurs only during early developmental stages, supporting the idea that maternal contribution of ift gene products masks the function of IFT proteins during initial development. In addition, the basal bodies in multiciliated cells of the pronephric duct in ift mutants were disorganized, with a pattern suggestive of defective planar cell polarity (PCP). Depletion of pk1, a core PCP component, similarly led to kidney cyst formation and basal body disorganization. Furthermore, we found that multiple ift genes genetically interact with pk1. Taken together, these data suggest that IFT proteins play a conserved role in cilia formation and planar cell polarity in zebrafish.
引用
收藏
页码:1326 / 1333
页数:8
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