MEIS-WNT5A axis regulates development of fourth ventricle
choroid plexus

J 2021

MEIS-WNT5A axis regulates development of fourth ventricle choroid plexus

KAISER, Karol, Ahram JANG, Petra KOMPANÍKOVÁ, Melody P. LUN, Jan PROCHAZKA et. al.

Basic information

Original name

MEIS-WNT5A axis regulates development of fourth ventricle choroid plexus

Authors

KAISER, Karol (703 Slovakia, guarantor, belonging to the institution), Ahram JANG, Petra KOMPANÍKOVÁ (703 Slovakia, belonging to the institution), Melody P. LUN, Jan PROCHAZKA, Ondrej MACHON, Neil DANI, Michaela PROCHAZKOVA, Benoit LAURENT, Daniel GYLLBORG, Renee VAN AMERONGEN, Ryann M. FAME, Suhasini GUPTA, Feizhen WU, Roger A. BARKER, Ivana BUKOVA, Radislav SEDLACEK, Zbynek KOZMIK, Ernest ARENAS, Maria K. LEHTINEN (203 Czech Republic) and Vítězslav BRYJA (203 Czech Republic, belonging to the institution)

Edition

Development, Company of Biologists Ltd, 2021, 0950-1991

Other information

Language

English

Type of outcome

Článek v odborném periodiku

Field of Study

10605 Developmental biology

Country of publisher

United Kingdom of Great Britain and Northern Ireland

Confidentiality degree

není předmětem státního či obchodního tajemství

References:

URL

Impact factor

Impact factor: 6.862

RIV identification code

RIV/00216224:14310/21:00123746

Organization unit

Faculty of Science

DOI

http://dx.doi.org/10.1242/dev.192054

UT WoS

000657670600005

Keywords in English

Choroid plexus; Epithelium; Meis1; Meis2; Morphogenesis; WNT5a

Abstract

V originále

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.

Links

LM2018129, research and development project

Name: Národní infrastruktura pro biologické a medicínské zobrazování Czech-BioImaging

Investor: Ministry of Education, Youth and Sports of the CR

Citovat

KAISER, Karol, Ahram JANG, Petra KOMPANÍKOVÁ, Melody P. LUN, Jan PROCHAZKA, Ondrej MACHON, Neil DANI, Michaela PROCHAZKOVA, Benoit LAURENT, Daniel GYLLBORG, Renee VAN AMERONGEN, Ryann M. FAME, Suhasini GUPTA, Feizhen WU, Roger A. BARKER, Ivana BUKOVA, Radislav SEDLACEK, Zbynek KOZMIK, Ernest ARENAS, Maria K. LEHTINEN and Vítězslav BRYJA. MEIS-WNT5A axis regulates development of fourth ventricle choroid plexus. Development. Company of Biologists Ltd, 2021, vol. 148, No 10, p. "dev192054", 17 pp. ISSN 0950-1991. Available from: https://dx.doi.org/10.1242/dev.192054.

@article{1821641,
   author = {Kaiser, Karol and Jang, Ahram and Kompaníková, Petra and Lun, Melody P. and Prochazka, Jan and Machon, Ondrej and Dani, Neil and Prochazkova, Michaela and Laurent, Benoit and Gyllborg, Daniel and van Amerongen, Renee and Fame, Ryann M. and Gupta, Suhasini and Wu, Feizhen and Barker, Roger A. and Bukova, Ivana and Sedlacek, Radislav and Kozmik, Zbynek and Arenas, Ernest and Lehtinen, Maria K. and Bryja, Vítězslav},
   article_number = {10},
   doi = {http://dx.doi.org/10.1242/dev.192054},
   keywords = {Choroid plexus; Epithelium; Meis1; Meis2; Morphogenesis; WNT5a},
   language = {eng},
   issn = {0950-1991},
   journal = {Development},
   title = {MEIS-WNT5A axis regulates development of fourth ventricle choroid plexus},
   url = {https://journals.biologists.com/dev/article/148/10/dev192054/268365/MEIS-WNT5A-axis-regulates-development-of-fourth},
   volume = {148},
   year = {2021}
}

TY  - JOUR
ID  - 1821641
AU  - Kaiser, Karol - Jang, Ahram - Kompaníková, Petra - Lun, Melody P. - Prochazka, Jan - Machon, Ondrej - Dani, Neil - Prochazkova, Michaela - Laurent, Benoit - Gyllborg, Daniel - van Amerongen, Renee - Fame, Ryann M. - Gupta, Suhasini - Wu, Feizhen - Barker, Roger A. - Bukova, Ivana - Sedlacek, Radislav - Kozmik, Zbynek - Arenas, Ernest - Lehtinen, Maria K. - Bryja, Vítězslav
PY  - 2021
TI  - MEIS-WNT5A axis regulates development of fourth ventricle choroid plexus
JF  - Development
VL  - 148
IS  - 10
SP  - "dev192054"
EP  - "dev192054"
PB  - Company of Biologists Ltd
SN  - 09501991
KW  - Choroid plexus
KW  - Epithelium
KW  - Meis1
KW  - Meis2
KW  - Morphogenesis
KW  - WNT5a
UR  - https://journals.biologists.com/dev/article/148/10/dev192054/268365/MEIS-WNT5A-axis-regulates-development-of-fourth
N2  - 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.
ER  -

KAISER, Karol, Ahram JANG, Petra KOMPANÍKOVÁ, Melody P. LUN, Jan PROCHAZKA, Ondrej MACHON, Neil DANI, Michaela PROCHAZKOVA, Benoit LAURENT, Daniel GYLLBORG, Renee VAN AMERONGEN, Ryann M. FAME, Suhasini GUPTA, Feizhen WU, Roger A. BARKER, Ivana BUKOVA, Radislav SEDLACEK, Zbynek KOZMIK, Ernest ARENAS, Maria K. LEHTINEN and Vítězslav BRYJA. MEIS-WNT5A axis regulates development of fourth ventricle choroid plexus. \textit{Development}. Company of Biologists Ltd, 2021, vol.~148, No~10, p.~''dev192054'', 17 pp. ISSN~0950-1991. Available from: https://dx.doi.org/10.1242/dev.192054.

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