Journal
DEVELOPMENT
Volume 132, Issue 8, Pages 1907-1921Publisher
COMPANY BIOLOGISTS LTD
DOI: 10.1242/dev.01772
Keywords
cilia; pronephros; Kupffer's vesicle; ependymal cell; spinal canal; kidney cyst; hydrocephalus; left-right asymmetry
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Funding
- NIDDK NIH HHS [T32 DK07545, P30 DK074038, DK53093, DK65655, DK54711] Funding Source: Medline
- NIGMS NIH HHS [5RO1 GM56211] Funding Source: Medline
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Cilia, as motile and sensory organelles, have been implicated in normal development, as well as diseases including cystic kidney disease, hydrocephalus and situs inversus. In kidney epithelia, cilia are proposed to be nonmotile sensory organelles, while in the mouse node, two cilia populations, motile and non-motile have been proposed to regulate situs. We show that cilia in the zebrafish larval kidney, the spinal cord and Kupffer's vesicle are motile, suggesting that fluid flow is a common feature of each of these organs. Disruption of cilia structure or motility resulted in pronephric cyst formation, hydrocephalus and left-right asymmetry defects. The data show that loss of fluid flow leads to fluid accumulation, which can account for organ distension pathologies in the kidney and brain. In Kupffer's vesicle, loss of flow is associated with loss of left-right patterning, indicating that the 'nodal flow' mechanism of generating situs is conserved in non-mammalian vertebrates.
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