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MEIS-WNT5A axis regulates development of fourth ventricle choroid plexus
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SYSNO ASEP 0554722 Document Type J - Journal Article R&D Document Type Journal Article Subsidiary J Článek ve WOS Title MEIS-WNT5A axis regulates development of fourth ventricle choroid plexus Author(s) Kaiser, K. (CZ)
Jang, A. (US)
Kompanikova, P. (CZ)
Lun, M.P. (US)
Procházka, Jan (UMG-J) ORCID
Machoň, Ondřej (UEM-P)
Dani, N. (US)
Procházková, Michaela (UMG-J)
Laurent, B. (CA)
Gyllborg, D. (SE)
van Amerongen, R. (NL)
Fame, R. (US)
Gupta, S. (US)
Wu, F. (CN)
Barker, R. (GB)
Buková, Ivana (UMG-J)
Sedláček, Radislav (UMG-J) RID
Kozmik, Zbyněk (UMG-J) RID
Arenas, E. (SE)
Lehtinen, M.K. (US)
Bryja, V. (CZ)Number of authors 21 Article number dev192054 Source Title Development. - : Company of Biologists - ISSN 0950-1991
Roč. 148, č. 10 (2021)Number of pages 18 s. Publication form Online - E Language eng - English Country GB - United Kingdom Keywords Choroid plexus ; Epithelium ; Meis1 ; Meis2 ; Morphogenesis ; WNT5a Subject RIV EB - Genetics ; Molecular Biology OECD category Developmental biology R&D Projects GA18-00514S GA ČR - Czech Science Foundation (CSF) GA18-20759S GA ČR - Czech Science Foundation (CSF) Method of publishing Limited access Institutional support UMG-J - RVO:68378050 ; UEM-P - RVO:68378041 UT WOS 000657670600005 DOI 10.1242/dev.192054 Annotation The choroid plexus (ChP) produces cerebrospinal fluid and forms an essential brain barrier. ChP tissues form in each brain ventricle, each one adopting a distinct shape, but remarkably little is known about the mechanisms underlying ChP development. Here, we show that epithelial WNT5A is crucial for determining fourth ventricle (4V) ChP morphogenesis and size in mouse. Systemic Wnt5a knockout, or forced Wnt5a overexpression beginning at embryonic day 10.5, profoundly reduced ChP size and development. However, Wnt5a expression was enriched in Foxj1-positive epithelial cells of 4V ChP plexus, and its conditional deletion in these cells affected 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 and 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 MEIS transcription factors as upstream regulators of Wnt5a in the 4V ChP epithelium. Workplace Institute of Molecular Genetics Contact Nikol Škňouřilová, nikol.sknourilova@img.cas.cz, Tel.: 241 063 217 Year of Publishing 2022 Electronic address https://journals.biologists.com/dev/article/148/10/dev192054/268365/MEIS-WNT5A-axis-regulates-development-of-fourth
Number of the records: 1