TY - JOUR
T1 - Chondroitin sulfate proteoglycan 4 regulates zebrafish body axis organization via Wnt/ planar cell polarity pathway
AU - Lee, Yen Hua
AU - Kawakami, Koichi
AU - HuangFu, Wei Chun
AU - Liu, I. Hsuan
N1 - Funding Information:
This work was financially supported by Ministry of Science and Technology, Taiwan (105-2628-B-002-005-MY4 to I-Hsuan Liu and 105-2917-I-002-033 to Yen-Hua Lee), Council of Agriculture, Taiwan (107AS-22.1.6-AD-U1(13) and 108AS-21.1.7-AD-U1(14) to I-Hsuan Liu) and National Taiwan University (107L7719 and 108L7706 to I-Hsuan Liu). The funders had no role in study design, data collection and analysis, decision to publish, or preparation of the manuscript. We would like to acknowledge Dr. Harry J. Mersmann and Dr. Simon Shu-Yu Wu for the reviewing and revising of this article.
Publisher Copyright:
© 2020 Lee et al. This is an open access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
PY - 2020/1/1
Y1 - 2020/1/1
N2 - Pericellular and extracellular proteoglycans play an important role in modulating morphogen gradients and signal transductions. Chondroitin sulfate proteoglycan 4 (Cspg4) is a membrane spanning proteoglycan expressed in immature progenitor cells and cancer cells. Cspg4 participates in cellular events such as proliferation, migration and signal transduction, and these events are generally important for embryo development. In this study, we characterized Cspg4 for its roles in zebrafish embryonic development. Our results demonstrated that cspg4 was maternally expressed from 0 to 3 hours post fertilization (hpf) and expressed in the anterior and posterior embryo end after 9 hpf. Knocking-down cspg4 resulted in a shorter anterior-posterior axis than control embryo, which could be rescued by co-injecting wnt11 mRNA suggesting that Cspg4 regulates body axis organization through modulating the Wnt/planar cell polarity signaling pathway. In addition, overexpressing cspg4 caused cyclopia. The Cspg4 transmembrane domain mutant embryo phenocopied the global overexpression of cspg4 mRNA and led to cyclopia with a very low penetrance. Our results demonstrated that the quantitatively and spatially accurate distribution of Cspg4 is critical for body axis and midline development during gastrulation.
AB - Pericellular and extracellular proteoglycans play an important role in modulating morphogen gradients and signal transductions. Chondroitin sulfate proteoglycan 4 (Cspg4) is a membrane spanning proteoglycan expressed in immature progenitor cells and cancer cells. Cspg4 participates in cellular events such as proliferation, migration and signal transduction, and these events are generally important for embryo development. In this study, we characterized Cspg4 for its roles in zebrafish embryonic development. Our results demonstrated that cspg4 was maternally expressed from 0 to 3 hours post fertilization (hpf) and expressed in the anterior and posterior embryo end after 9 hpf. Knocking-down cspg4 resulted in a shorter anterior-posterior axis than control embryo, which could be rescued by co-injecting wnt11 mRNA suggesting that Cspg4 regulates body axis organization through modulating the Wnt/planar cell polarity signaling pathway. In addition, overexpressing cspg4 caused cyclopia. The Cspg4 transmembrane domain mutant embryo phenocopied the global overexpression of cspg4 mRNA and led to cyclopia with a very low penetrance. Our results demonstrated that the quantitatively and spatially accurate distribution of Cspg4 is critical for body axis and midline development during gastrulation.
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U2 - 10.1371/journal.pone.0230943
DO - 10.1371/journal.pone.0230943
M3 - Article
AN - SCOPUS:85082802290
SN - 1932-6203
VL - 15
JO - PLoS ONE
JF - PLoS ONE
IS - 4
M1 - e0230943
ER -
