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Evaluation of Planar-Cell-Polarity Phenotypes in Ciliopathy Mouse Mutant Cochlea
Published on: February 21, 2016
Ciliary and non-ciliary expression and function of PACRG during vertebrate development
Thomas Thumberger1, Cathrin Hagenlocher, Matthias Tisler
1Institute of Zoology, Working group Embryology, University of Hohenheim, Garbenstraße 30, Stuttgart, 70593, Germany. martin.blum@uni-hohenheim.de.
Cilia
|January 29, 2013
Summary
Park2-co-regulated gene (PACRG) is essential for cilia function and embryonic development. Loss of PACRG in Xenopus causes defects in left-right axis formation and neural tube closure, highlighting its critical role.
Area of Science:
- Developmental Biology
- Cell Biology
- Genetics
Background:
- Park2-co-regulated gene (PACRG) is conserved across species and associated with flagella, impacting their formation.
- PACRG is crucial for spermatogenesis in mice.
- Its role in vertebrate embryogenesis and left-right (LR) axis specification via cilia-driven flow was unexplored.
Purpose of the Study:
- To examine PACRG expression patterns during vertebrate embryogenesis.
- To determine if PACRG is required for left-right (LR) axis specification in Xenopus laevis, particularly concerning cilia-driven leftward flow.
Main Methods:
- Cloned PACRG cDNAs and analyzed expression in Xenopus, mouse, rabbit, and zebrafish embryos.
- Utilized antisense morpholino oligonucleotide (MO) mediated gene knockdown in Xenopus.
- Investigated LR development using timelapse videography, scanning electron microscopy (SEM), and whole-mount in situ hybridization.
Main Results:
- PACRG mRNA was detected in ciliated cells and tissues, including the brain, across vertebrates.
- PACRG localized to epithelia crucial for leftward flow (GRP in Xenopus, PNC in mammals, KV in zebrafish).
- Loss-of-function experiments in Xenopus led to dose-dependent defects in LR axis formation, neural tube closure, and gastrulation.
Conclusions:
- PACRG protein is a novel and essential component of cilia in Xenopus.
- PACRG exhibits both ciliary and non-ciliary functions during embryonic development.
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