Synergistic roles of Wnt modulators R-spondin2 and R-spondin3 in craniofacial morphogenesis and dental development

Abstract Wnt signaling plays a critical role in craniofacial patterning, as well as tooth and bone development. Rspo2 and Rspo3 are key regulators of Wnt signaling. However, their coordinated function and relative requirement in craniofacial development and odontogensis are poorly understood. We sho...

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Autores principales:	Nora Alhazmi, Shannon H. Carroll, Kenta Kawasaki, Katherine C. Woronowicz, Shawn A. Hallett, Claudio Macias Trevino, Edward B. Li, Roland Baron, Francesca Gori, Pamela C. Yelick, Matthew P. Harris, Eric C. Liao
Formato:	article
Lenguaje:	EN
Publicado:	Nature Portfolio 2021
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/2322517fdbda474e8099cd2cb355bd86
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spelling	oai:doaj.org-article:2322517fdbda474e8099cd2cb355bd862021-12-02T13:30:37ZSynergistic roles of Wnt modulators R-spondin2 and R-spondin3 in craniofacial morphogenesis and dental development10.1038/s41598-021-85415-y2045-2322https://doaj.org/article/2322517fdbda474e8099cd2cb355bd862021-03-01T00:00:00Zhttps://doi.org/10.1038/s41598-021-85415-yhttps://doaj.org/toc/2045-2322Abstract Wnt signaling plays a critical role in craniofacial patterning, as well as tooth and bone development. Rspo2 and Rspo3 are key regulators of Wnt signaling. However, their coordinated function and relative requirement in craniofacial development and odontogensis are poorly understood. We showed that in zebrafish rspo2 and rspo3 are both expressed in osteoprogenitors in the embryonic craniofacial skeleton. This is in contrast to mouse development, where Rspo3 is expressed in osteoprogenitors while Rspo2 expression is not observed. In zebrafish, rspo2 and rspo3 are broadly expressed in the pulp, odontoblasts and epithelial crypts. However, in the developing molars of the mouse, Rspo3 is largely expressed in the dental follicle and alveolar mesenchyme while Rspo2 expression is restricted to the tooth germ. While Rspo3 ablation in the mouse is embryonic lethal, zebrafish rspo3-/- mutants are viable with modest decrease in Meckel’s cartilage rostral length. However, compound disruption of rspo3 and rspo2 revealed synergistic roles of these genes in cartilage morphogenesis, fin development, and pharyngeal tooth development. Adult rspo3 −/− zebrafish mutants exhibit a dysmorphic cranial skeleton and decreased average tooth number. This study highlights the differential functions of Rspo2 and Rspo3 in dentocranial morphogenesis in zebrafish and in mouse.Nora AlhazmiShannon H. CarrollKenta KawasakiKatherine C. WoronowiczShawn A. HallettClaudio Macias TrevinoEdward B. LiRoland BaronFrancesca GoriPamela C. YelickMatthew P. HarrisEric C. LiaoNature PortfolioarticleMedicineRScienceQENScientific Reports, Vol 11, Iss 1, Pp 1-14 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Nora Alhazmi Shannon H. Carroll Kenta Kawasaki Katherine C. Woronowicz Shawn A. Hallett Claudio Macias Trevino Edward B. Li Roland Baron Francesca Gori Pamela C. Yelick Matthew P. Harris Eric C. Liao Synergistic roles of Wnt modulators R-spondin2 and R-spondin3 in craniofacial morphogenesis and dental development
description	Abstract Wnt signaling plays a critical role in craniofacial patterning, as well as tooth and bone development. Rspo2 and Rspo3 are key regulators of Wnt signaling. However, their coordinated function and relative requirement in craniofacial development and odontogensis are poorly understood. We showed that in zebrafish rspo2 and rspo3 are both expressed in osteoprogenitors in the embryonic craniofacial skeleton. This is in contrast to mouse development, where Rspo3 is expressed in osteoprogenitors while Rspo2 expression is not observed. In zebrafish, rspo2 and rspo3 are broadly expressed in the pulp, odontoblasts and epithelial crypts. However, in the developing molars of the mouse, Rspo3 is largely expressed in the dental follicle and alveolar mesenchyme while Rspo2 expression is restricted to the tooth germ. While Rspo3 ablation in the mouse is embryonic lethal, zebrafish rspo3-/- mutants are viable with modest decrease in Meckel’s cartilage rostral length. However, compound disruption of rspo3 and rspo2 revealed synergistic roles of these genes in cartilage morphogenesis, fin development, and pharyngeal tooth development. Adult rspo3 −/− zebrafish mutants exhibit a dysmorphic cranial skeleton and decreased average tooth number. This study highlights the differential functions of Rspo2 and Rspo3 in dentocranial morphogenesis in zebrafish and in mouse.
format	article
author	Nora Alhazmi Shannon H. Carroll Kenta Kawasaki Katherine C. Woronowicz Shawn A. Hallett Claudio Macias Trevino Edward B. Li Roland Baron Francesca Gori Pamela C. Yelick Matthew P. Harris Eric C. Liao
author_facet	Nora Alhazmi Shannon H. Carroll Kenta Kawasaki Katherine C. Woronowicz Shawn A. Hallett Claudio Macias Trevino Edward B. Li Roland Baron Francesca Gori Pamela C. Yelick Matthew P. Harris Eric C. Liao
author_sort	Nora Alhazmi
title	Synergistic roles of Wnt modulators R-spondin2 and R-spondin3 in craniofacial morphogenesis and dental development
title_short	Synergistic roles of Wnt modulators R-spondin2 and R-spondin3 in craniofacial morphogenesis and dental development
title_full	Synergistic roles of Wnt modulators R-spondin2 and R-spondin3 in craniofacial morphogenesis and dental development
title_fullStr	Synergistic roles of Wnt modulators R-spondin2 and R-spondin3 in craniofacial morphogenesis and dental development
title_full_unstemmed	Synergistic roles of Wnt modulators R-spondin2 and R-spondin3 in craniofacial morphogenesis and dental development
title_sort	synergistic roles of wnt modulators r-spondin2 and r-spondin3 in craniofacial morphogenesis and dental development
publisher	Nature Portfolio
publishDate	2021
url	https://doaj.org/article/2322517fdbda474e8099cd2cb355bd86
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Synergistic roles of Wnt modulators R-spondin2 and R-spondin3 in craniofacial morphogenesis and dental development

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