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
-
- Animals
- Polycystic Kidney Diseases*/genetics
- Polycystic Kidney Diseases*/metabolism
- Polycystic Kidney Diseases*/pathology
- Gene Deletion*
- Zebrafish*/genetics
- Zebrafish*/metabolism
- Disease Models, Animal
- Zebrafish Proteins*/genetics
- Zebrafish Proteins*/metabolism
- PubMed
- 30254740 Full text @ F1000Res
Citation
Molinari, E., Ramsbottom, S.A., Sammut, V., Hughes, F.E.P., Sayer, J.A. (2018) Using zebrafish to study the function of nephronophthisis and related ciliopathy genes. F1000Research. 7:1133.
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
Expression
Phenotype
Mutations / Transgenics
Human Disease / Model
Sequence Targeting Reagents
Fish
Orthology
Engineered Foreign Genes
Mapping