ZFIN ID: ZDB-PUB-101011-17
Notch signalling regulates left-right asymmetry through ciliary length control
Lopes, S.S., Lourenço, R., Pacheco, L., Moreno, N., Kreiling, J., and Saude, L.
Date: 2010
Source: Development (Cambridge, England)   137(21): 3625-3632 (Journal)
Registered Authors: Lopes, Susana, Lourenço, Raquel, Pacheco, Luis, Saude, Leonor
Keywords: Notch, Cilia, Left-right, Zebrafish
MeSH Terms:
  • Animals
  • Animals, Genetically Modified
  • Body Patterning/genetics*
  • Cilia/metabolism
  • Cilia/physiology*
  • Embryo, Nonmammalian
  • Gene Expression Regulation, Developmental
  • Intestines/embryology
  • Intestines/metabolism
  • Nerve Tissue Proteins/genetics
  • Nerve Tissue Proteins/metabolism
  • Nerve Tissue Proteins/physiology
  • Organ Size/genetics
  • Protein Binding
  • Receptors, Notch/genetics
  • Receptors, Notch/metabolism
  • Receptors, Notch/physiology*
  • Signal Transduction/genetics
  • Signal Transduction/physiology
  • Water-Electrolyte Balance/genetics
  • Water-Electrolyte Balance/physiology
  • Zebrafish/embryology
  • Zebrafish/genetics
  • Zebrafish/physiology
  • Zebrafish Proteins/genetics
  • Zebrafish Proteins/metabolism
  • Zebrafish Proteins/physiology
PubMed: 20876649 Full text @ Development
The importance of cilia in embryonic development and adult physiology is emphasized by human ciliopathies. Despite its relevance, molecular signalling pathways behind cilia formation are poorly understood. We show that Notch signalling is a key pathway for cilia length control. In deltaD zebrafish mutants, cilia length is reduced in Kupffers vesicle and can be rescued by the ciliogenic factor foxj1a. Conversely, cilia length increases when Notch signalling is hyperactivated. Short cilia found in deltaD mutants reduce the fluid flow velocity inside Kupffers vesicle, thus compromising the asymmetric expression of the flow sensor charon. Notch signalling brings together ciliary length control and fluid flow hydrodynamics with transcriptional activation of laterality genes. In addition, our deltaD mutant analysis discloses an uncoupling between gut and heart laterality.