PUBLICATION

Using zebrafish to study the function of nephronophthisis and related ciliopathy genes

Authors
Molinari, E., Ramsbottom, S.A., Sammut, V., Hughes, F.E.P., Sayer, J.A.
ID
ZDB-PUB-180927-14
Date
2018
Source
F1000Research   7: 1133 (Journal)
Registered Authors
Sayer, John A.
Keywords
Kupffer’s vesicle; acetylated alpha-tubulin, primary cilia, somite
MeSH Terms
  • Disease Models, Animal
  • Polycystic Kidney Diseases*/genetics
  • Polycystic Kidney Diseases*/metabolism
  • Polycystic Kidney Diseases*/pathology
  • Zebrafish*/genetics
  • Zebrafish*/metabolism
  • Gene Deletion*
  • Animals
  • Zebrafish Proteins*/genetics
  • Zebrafish Proteins*/metabolism
(all 10)
PubMed
30254740 Full text @ F1000Res
Abstract
Zebrafish are a valuable vertebrate model in which to study development and characterize genes involved in cystic kidney disease. Zebrafish embryos and larvae are transparent, allowing non-invasive imaging during their rapid development, which takes place over the first 72 hours post fertilisation. Gene-specific knockdown of nephronophthisis-associated genes leads to ciliary phenotypes which can be assessed in various developmental structures. Here we describe in detail the methods used for imaging cilia within Kupffer's vesicle to assess nephronophthisis and related ciliopathy phenotypes.
Genes / Markers
Figures
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Expression
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Phenotype
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Mutations / Transgenics
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Human Disease / Model
Human Disease Fish Conditions Evidence
ciliopathyTAS
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Sequence Targeting Reagents
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Fish
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Antibodies
Orthology
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Engineered Foreign Genes
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Mapping
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