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Evaluation of Planar-Cell-Polarity Phenotypes in Ciliopathy Mouse Mutant Cochlea
Published on: February 21, 2016
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La-related protein 6 controls ciliated cell differentiation.
Zarko Manojlovic1,2, Ryan Earwood1, Akiko Kato1
1Department of Biomedical Sciences, Florida State University College of Medicine, 1115W. Call Street, Tallahassee, FL 32306-4300 USA.
Cilia
|March 28, 2017
Summary
La-related protein 6 (LARP6) is crucial for ciliated cell differentiation during development. It regulates the expression of cilia-related genes, distinct from its known role in collagen mRNA translation.
Area of Science:
- Developmental Biology
- Cell Biology
- Molecular Biology
Background:
- La-related protein 6 (LARP6) is an evolutionarily conserved RNA-binding protein.
- While known to regulate type I collagen mRNA translation, other functions and targets of LARP6 remain largely unknown.
- This study aimed to uncover additional functions of LARP6 and assess its developmental importance.
Purpose of the Study:
- To investigate the role of LARP6 in embryonic development.
- To elucidate the molecular mechanisms by which LARP6 influences ciliogenesis.
- To evaluate the significance of LARP6 in ciliated cell differentiation.
Main Methods:
- LARP6 was depleted in *Xenopus* embryos using Morpholino Oligos.
- Embryonic phenotypes and ciliary structures of LARP6-depleted embryos (morphants) were analyzed.
- Expression of key ciliogenesis genes was assessed using RT-PCR and whole-mount in situ hybridization (WISH).
Main Results:
- LARP6 depletion caused neural tube closure defects and lack of cilia in the neural tube and epidermis.
- These defects were linked to the absence of basal bodies, essential for ciliary assembly.
- Partial restoration of multi-ciliated cells (MCCs) was observed upon manipulating *mcidas* expression, a gene regulating centriole formation.
- LARP6 appears to control *mcidas* expression independently of the Notch signaling pathway.
Conclusions:
- LARP6 plays a vital role in ciliated cell differentiation during development.
- This function is mediated by controlling the expression of cilia-related genes, including *mcidas*.
- The identified role of LARP6 in ciliogenesis is distinct from its previously established function in collagen mRNA translation.
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