PUBLICATION

Disruption of planar cell polarity activity leads to developmental biliary defects

Authors
Cui, S., Capecci, L.M., and Matthews, R.P.
ID
ZDB-PUB-110119-7
Date
2011
Source
Developmental Biology   351(2): 229-241 (Journal)
Registered Authors
Matthews, Randy
Keywords
none
MeSH Terms
  • Zebrafish Proteins/physiology*
  • Hepatocyte Nuclear Factor 1-beta/physiology*
  • Cell Polarity*
  • Animals
  • Zebrafish/embryology*
  • Bile Ducts/abnormalities*
  • Bile Ducts/cytology
  • Carrier Proteins/physiology*
  • LIM Domain Proteins
  • Adaptor Proteins, Signal Transducing
  • Liver/metabolism
  • Gene Expression Regulation, Developmental
(all 12)
PubMed
21215262 Full text @ Dev. Biol.
Abstract
Planar cell polarity (PCP) establishes polarity within an epithelial sheet. Defects in PCP are associated with developmental defects involving directional cell growth, including defects in kidney tubule elongation that lead to formation of kidney cysts. Given the strong association between kidney cyst formation and developmental biliary defects in patients and in animal models, we investigated the importance of PCP in biliary development. Here we report that in zebrafish, morpholino antisense oligonucleotide-mediated knockdown of PCP genes including prickle-1a (pk1a) led to developmental biliary abnormalities, as well as localization defects of the liver and other digestive organs. The defects in biliary development appear to be mediated via downstream PCP targets such as Rho kinase, Jun kinase (JNK), and both actin and microtubule components of the cytoskeleton. Knockdown of pk1a led to decreased expression of vhnf1, a homeodomain gene previously shown to be involved in biliary development and in kidney cyst formation; forced expression of vhnf1 mRNA led to rescue of the pk1a morphant phenotype. Our results demonstrate that PCP plays an important role in vertebrate biliary development, interacting with other factors known to be involved in biliary morphogenesis.
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Sequence Targeting Reagents
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Fish
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Name Type Antigen Genes Isotypes Host Organism
Ab1-krt18monoclonal
    IgG1Mouse
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