Skip to main content
bioRxiv
  • Home
  • About
  • Submit
  • ALERTS / RSS
Advanced Search
New Results

MEIS-WNT5A axis regulates development of 4th ventricle choroid plexus

View ORCID ProfileKarol Kaiser, View ORCID ProfileAhram Jang, Melody P. Lun, View ORCID ProfileJan Procházka, View ORCID ProfileOndrej Machon, View ORCID ProfileMichaela Procházková, Benoit Laurent, View ORCID ProfileDaniel Gyllborg, View ORCID ProfileRenée van Amerongen, View ORCID ProfilePetra Kompaníková, View ORCID ProfileFeizhen Wu, View ORCID ProfileRoger A. Barker, Ivana Uramová, View ORCID ProfileRadislav Sedláček, View ORCID ProfileZbyněk Kozmík, View ORCID ProfileErnest Arenas, View ORCID ProfileMaria K. Lehtinen, View ORCID ProfileVítězslav Bryja
doi: https://doi.org/10.1101/2020.05.07.082370
Karol Kaiser
1Department of Experimental Biology, Faculty of Science, Masaryk University, Brno, Czech Republic
  • Find this author on Google Scholar
  • Find this author on PubMed
  • Search for this author on this site
  • ORCID record for Karol Kaiser
Ahram Jang
2Department of Pathology, Boston Children’s Hospital, Boston, Massachusetts, USA
  • Find this author on Google Scholar
  • Find this author on PubMed
  • Search for this author on this site
  • ORCID record for Ahram Jang
Melody P. Lun
2Department of Pathology, Boston Children’s Hospital, Boston, Massachusetts, USA
  • Find this author on Google Scholar
  • Find this author on PubMed
  • Search for this author on this site
Jan Procházka
3Czech Centre for Phenogenomics and Laboratory of Transgenic Models of Diseases, Institute of Molecular Genetics of the CAS, Czech Republic
  • Find this author on Google Scholar
  • Find this author on PubMed
  • Search for this author on this site
  • ORCID record for Jan Procházka
Ondrej Machon
4Department of Developmental Biology, Institute of Experimental Medicine of the CAS, Prague, Czech Republic
  • Find this author on Google Scholar
  • Find this author on PubMed
  • Search for this author on this site
  • ORCID record for Ondrej Machon
Michaela Procházková
3Czech Centre for Phenogenomics and Laboratory of Transgenic Models of Diseases, Institute of Molecular Genetics of the CAS, Czech Republic
  • Find this author on Google Scholar
  • Find this author on PubMed
  • Search for this author on this site
  • ORCID record for Michaela Procházková
Benoit Laurent
5Research Center on Aging, CIUSSS de l’Estrie - CHUS, Sherbrooke, QC, Canada
6Department of Biochemistry and Functional Genomics, Faculty of Medicine and Health Sciences, Université de Sherbrooke, Sherbrooke, QC, Canada
  • Find this author on Google Scholar
  • Find this author on PubMed
  • Search for this author on this site
Daniel Gyllborg
7Science for Life Laboratory, Department of Biochemistry and Biophysics, Stockholm University, Solna, Sweden
  • Find this author on Google Scholar
  • Find this author on PubMed
  • Search for this author on this site
  • ORCID record for Daniel Gyllborg
Renée van Amerongen
8Swammerdam Institute for Life Sciences, Faculty of Science, University of Amsterdam, Netherlands
  • Find this author on Google Scholar
  • Find this author on PubMed
  • Search for this author on this site
  • ORCID record for Renée van Amerongen
Petra Kompaníková
1Department of Experimental Biology, Faculty of Science, Masaryk University, Brno, Czech Republic
  • Find this author on Google Scholar
  • Find this author on PubMed
  • Search for this author on this site
  • ORCID record for Petra Kompaníková
Feizhen Wu
9Shanghai Institute of Cardiovascular Diseases, Zhongshan Hospital, and Institutes of Biomedical Sciences, Fudan University, Shanghai, China
  • Find this author on Google Scholar
  • Find this author on PubMed
  • Search for this author on this site
  • ORCID record for Feizhen Wu
Roger A. Barker
10John van Geest Centre for Brain Repair and WT-MRc Cambridge Stem Cell Centre, University of Cambridge, Cambridge, UK
  • Find this author on Google Scholar
  • Find this author on PubMed
  • Search for this author on this site
  • ORCID record for Roger A. Barker
Ivana Uramová
3Czech Centre for Phenogenomics and Laboratory of Transgenic Models of Diseases, Institute of Molecular Genetics of the CAS, Czech Republic
  • Find this author on Google Scholar
  • Find this author on PubMed
  • Search for this author on this site
Radislav Sedláček
3Czech Centre for Phenogenomics and Laboratory of Transgenic Models of Diseases, Institute of Molecular Genetics of the CAS, Czech Republic
  • Find this author on Google Scholar
  • Find this author on PubMed
  • Search for this author on this site
  • ORCID record for Radislav Sedláček
Zbyněk Kozmík
4Department of Developmental Biology, Institute of Experimental Medicine of the CAS, Prague, Czech Republic
  • Find this author on Google Scholar
  • Find this author on PubMed
  • Search for this author on this site
  • ORCID record for Zbyněk Kozmík
Ernest Arenas
11Department of Medical Biochemistry and Biophysics, Karolinska Institutet, Stockholm, Sweden
  • Find this author on Google Scholar
  • Find this author on PubMed
  • Search for this author on this site
  • ORCID record for Ernest Arenas
Maria K. Lehtinen
2Department of Pathology, Boston Children’s Hospital, Boston, Massachusetts, USA
  • Find this author on Google Scholar
  • Find this author on PubMed
  • Search for this author on this site
  • ORCID record for Maria K. Lehtinen
  • For correspondence: maria.lehtinen@childrens.harvard.edu bryja@sci.muni.cz
Vítězslav Bryja
1Department of Experimental Biology, Faculty of Science, Masaryk University, Brno, Czech Republic
  • Find this author on Google Scholar
  • Find this author on PubMed
  • Search for this author on this site
  • ORCID record for Vítězslav Bryja
  • For correspondence: maria.lehtinen@childrens.harvard.edu bryja@sci.muni.cz
  • Abstract
  • Full Text
  • Info/History
  • Metrics
  • Preview PDF
Loading

ABSTRACT

The choroid plexus (ChP) produces cerebrospinal fluid and forms a critical barrier between the brain and the circulation. While the ChP forms in each brain ventricle, it adopts a different shape in each one and remarkably little is known about the mechanisms underlying its development. Here, we show that epithelial WNT5A is critical for determining fourth ventricle (4V) ChP morphogenesis and size. Systemic Wnt5a knockout, or forced WNT5A overexpression beginning at E10.5, profoundly reduced the size and development of ChP in all ventricles. However, conditional deletion of Wnt5a expression in Foxj1-expressing epithelial cells affected only the branched, villous morphology of the 4V ChP. We found that WNT5A was enriched in epithelial cells localized to the distal tips of 4V ChP villi, where WNT5A acted locally to activate non-canonical Wnt signaling via Ror1/Ror2 receptors. During 4V ChP development, MEIS1 bound to the proximal Wnt5a promoter, and gain- and loss-of-function approaches demonstrated that MEIS1 regulated Wnt5a expression. Collectively, our findings demonstrate a dual function of WNT5A in ChP development and identify MEIS1 and MEIS2 as upstream regulators of Wnt5a in the 4V ChP epithelium.

Competing Interest Statement

The authors have declared no competing interest.

Copyright 
The copyright holder for this preprint is the author/funder, who has granted bioRxiv a license to display the preprint in perpetuity. All rights reserved. No reuse allowed without permission.
Back to top
PreviousNext
Posted May 07, 2020.
Download PDF
Email

Thank you for your interest in spreading the word about bioRxiv.

NOTE: Your email address is requested solely to identify you as the sender of this article.

Enter multiple addresses on separate lines or separate them with commas.
MEIS-WNT5A axis regulates development of 4th ventricle choroid plexus
(Your Name) has forwarded a page to you from bioRxiv
(Your Name) thought you would like to see this page from the bioRxiv website.
CAPTCHA
This question is for testing whether or not you are a human visitor and to prevent automated spam submissions.
Share
MEIS-WNT5A axis regulates development of 4th ventricle choroid plexus
Karol Kaiser, Ahram Jang, Melody P. Lun, Jan Procházka, Ondrej Machon, Michaela Procházková, Benoit Laurent, Daniel Gyllborg, Renée van Amerongen, Petra Kompaníková, Feizhen Wu, Roger A. Barker, Ivana Uramová, Radislav Sedláček, Zbyněk Kozmík, Ernest Arenas, Maria K. Lehtinen, Vítězslav Bryja
bioRxiv 2020.05.07.082370; doi: https://doi.org/10.1101/2020.05.07.082370
Digg logo Reddit logo Twitter logo Facebook logo Google logo LinkedIn logo Mendeley logo
Citation Tools
MEIS-WNT5A axis regulates development of 4th ventricle choroid plexus
Karol Kaiser, Ahram Jang, Melody P. Lun, Jan Procházka, Ondrej Machon, Michaela Procházková, Benoit Laurent, Daniel Gyllborg, Renée van Amerongen, Petra Kompaníková, Feizhen Wu, Roger A. Barker, Ivana Uramová, Radislav Sedláček, Zbyněk Kozmík, Ernest Arenas, Maria K. Lehtinen, Vítězslav Bryja
bioRxiv 2020.05.07.082370; doi: https://doi.org/10.1101/2020.05.07.082370

Citation Manager Formats

  • BibTeX
  • Bookends
  • EasyBib
  • EndNote (tagged)
  • EndNote 8 (xml)
  • Medlars
  • Mendeley
  • Papers
  • RefWorks Tagged
  • Ref Manager
  • RIS
  • Zotero
  • Tweet Widget
  • Facebook Like
  • Google Plus One
Subject Areas
All Articles
  • Animal Behavior and Cognition (3479)
  • Biochemistry (7318)
  • Bioengineering (5296)
  • Bioinformatics (20197)
  • Biophysics (9976)
  • Cancer Biology (7703)
  • Cell Biology (11250)
  • Clinical Trials (138)
  • Developmental Biology (6417)
  • Ecology (9916)
  • Epidemiology (2065)
  • Evolutionary Biology (13280)
  • Genetics (9352)
  • Genomics (12554)
  • Immunology (7674)
  • Microbiology (18939)
  • Molecular Biology (7417)
  • Neuroscience (40891)
  • Paleontology (298)
  • Pathology (1226)
  • Pharmacology and Toxicology (2126)
  • Physiology (3140)
  • Plant Biology (6838)
  • Scientific Communication and Education (1270)
  • Synthetic Biology (1891)
  • Systems Biology (5296)
  • Zoology (1085)