MEIS-WNT5A axis regulates development of fourth ventricle choroid plexus

0554722 - ÚMG 2022 RIV GB eng J - Článek v odborném periodiku
Kaiser, K. - Jang, A. - Kompanikova, P. - Lun, M.P. - Procházka, Jan - Machoň, Ondřej - Dani, N. - Procházková, Michaela - Laurent, B. - Gyllborg, D. - van Amerongen, R. - Fame, R. - Gupta, S. - Wu, F. - Barker, R. - Buková, Ivana - Sedláček, Radislav - Kozmik, Zbyněk - Arenas, E. - Lehtinen, M.K. - Bryja, V.
MEIS-WNT5A axis regulates development of fourth ventricle choroid plexus.
Development. Roč. 148, č. 10 (2021), č. článku dev192054. ISSN 0950-1991. E-ISSN 1477-9129
Grant CEP: GA ČR(CZ) GA18-00514S; GA ČR(CZ) GA18-20759S
Institucionální podpora: RVO:68378050 ; RVO:68378041
Klíčová slova: Choroid plexus * Epithelium * Meis1 * Meis2 * Morphogenesis * WNT5a
Obor OECD: Developmental biology; Developmental biology (UEM-P)
Impakt faktor: 6.862, rok: 2021
Způsob publikování: Omezený přístup
https://journals.biologists.com/dev/article/148/10/dev192054/268365/MEIS-WNT5A-axis-regulates-development-of-fourth

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.
Trvalý link: http://hdl.handle.net/11104/0329384