Runx2-Twist1 interaction coordinates cranial neural crest guidance of soft palate myogenesis

Cranial neural crest (CNC) cells give rise to bone, cartilage, tendons, and ligaments of the vertebrate craniofacial musculoskeletal complex, as well as regulate mesoderm-derived craniofacial muscle development through cell-cell interactions. Using the mouse soft palate as a model, we performed an u...

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Main Authors:	Xia Han, Jifan Feng, Tingwei Guo, Yong-Hwee Eddie Loh, Yuan Yuan, Thach-Vu Ho, Courtney Kyeong Cho, Jingyuan Li, Junjun Jing, Eva Janeckova, Jinzhi He, Fei Pei, Jing Bi, Brian Song, Yang Chai
Format:	Article
Language:	English
Published:	eLife Sciences Publications Ltd 2021-01-01
Series:	eLife
Subjects:	cleft palate Runx2 cell-cell interaction muscle development cranial neural crest cells
Online Access:	https://elifesciences.org/articles/62387

id	doaj-a213bfe478cd4f948d7dd30a98f531d5
record_format	Article
spelling	doaj-a213bfe478cd4f948d7dd30a98f531d52021-05-05T22:42:42ZengeLife Sciences Publications LtdeLife2050-084X2021-01-011010.7554/eLife.62387Runx2-Twist1 interaction coordinates cranial neural crest guidance of soft palate myogenesisXia Han0Jifan Feng1https://orcid.org/0000-0002-9944-2604Tingwei Guo2Yong-Hwee Eddie Loh3Yuan Yuan4https://orcid.org/0000-0002-7742-9433Thach-Vu Ho5https://orcid.org/0000-0001-6293-4739Courtney Kyeong Cho6Jingyuan Li7Junjun Jing8https://orcid.org/0000-0001-5745-5207Eva Janeckova9Jinzhi He10Fei Pei11Jing Bi12Brian Song13Yang Chai14https://orcid.org/0000-0003-2477-7247Center for Craniofacial Molecular Biology, University of Southern California, Los Angeles, Los Angeles, United StatesCenter for Craniofacial Molecular Biology, University of Southern California, Los Angeles, Los Angeles, United StatesCenter for Craniofacial Molecular Biology, University of Southern California, Los Angeles, Los Angeles, United StatesUSC Libraries Bioinformatics Services, University of Southern California, Los Angeles, Los Angeles, United StatesCenter for Craniofacial Molecular Biology, University of Southern California, Los Angeles, Los Angeles, United StatesCenter for Craniofacial Molecular Biology, University of Southern California, Los Angeles, Los Angeles, United StatesCenter for Craniofacial Molecular Biology, University of Southern California, Los Angeles, Los Angeles, United StatesCenter for Craniofacial Molecular Biology, University of Southern California, Los Angeles, Los Angeles, United StatesCenter for Craniofacial Molecular Biology, University of Southern California, Los Angeles, Los Angeles, United StatesCenter for Craniofacial Molecular Biology, University of Southern California, Los Angeles, Los Angeles, United StatesCenter for Craniofacial Molecular Biology, University of Southern California, Los Angeles, Los Angeles, United StatesCenter for Craniofacial Molecular Biology, University of Southern California, Los Angeles, Los Angeles, United StatesCenter for Craniofacial Molecular Biology, University of Southern California, Los Angeles, Los Angeles, United StatesCenter for Craniofacial Molecular Biology, University of Southern California, Los Angeles, Los Angeles, United StatesCenter for Craniofacial Molecular Biology, University of Southern California, Los Angeles, Los Angeles, United StatesCranial neural crest (CNC) cells give rise to bone, cartilage, tendons, and ligaments of the vertebrate craniofacial musculoskeletal complex, as well as regulate mesoderm-derived craniofacial muscle development through cell-cell interactions. Using the mouse soft palate as a model, we performed an unbiased single-cell RNA-seq analysis to investigate the heterogeneity and lineage commitment of CNC derivatives during craniofacial muscle development. We show that Runx2, a known osteogenic regulator, is expressed in the CNC-derived perimysial and progenitor populations. Loss of Runx2 in CNC-derivatives results in reduced expression of perimysial markers (Aldh1a2 and Hic1) as well as soft palate muscle defects in Osr2-Cre;Runx2fl/fl mice. We further reveal that Runx2 maintains perimysial marker expression through suppressing Twist1, and that myogenesis is restored in Osr2-Cre;Runx2fl/fl;Twist1fl/+ mice. Collectively, our findings highlight the roles of Runx2, Twist1, and their interaction in regulating the fate of CNC-derived cells as they guide craniofacial muscle development through cell-cell interactions.https://elifesciences.org/articles/62387cleft palateRunx2cell-cell interactionmuscle developmentcranial neural crest cells
collection	DOAJ
language	English
format	Article
sources	DOAJ
author	Xia Han Jifan Feng Tingwei Guo Yong-Hwee Eddie Loh Yuan Yuan Thach-Vu Ho Courtney Kyeong Cho Jingyuan Li Junjun Jing Eva Janeckova Jinzhi He Fei Pei Jing Bi Brian Song Yang Chai
spellingShingle	Xia Han Jifan Feng Tingwei Guo Yong-Hwee Eddie Loh Yuan Yuan Thach-Vu Ho Courtney Kyeong Cho Jingyuan Li Junjun Jing Eva Janeckova Jinzhi He Fei Pei Jing Bi Brian Song Yang Chai Runx2-Twist1 interaction coordinates cranial neural crest guidance of soft palate myogenesis eLife cleft palate Runx2 cell-cell interaction muscle development cranial neural crest cells
author_facet	Xia Han Jifan Feng Tingwei Guo Yong-Hwee Eddie Loh Yuan Yuan Thach-Vu Ho Courtney Kyeong Cho Jingyuan Li Junjun Jing Eva Janeckova Jinzhi He Fei Pei Jing Bi Brian Song Yang Chai
author_sort	Xia Han
title	Runx2-Twist1 interaction coordinates cranial neural crest guidance of soft palate myogenesis
title_short	Runx2-Twist1 interaction coordinates cranial neural crest guidance of soft palate myogenesis
title_full	Runx2-Twist1 interaction coordinates cranial neural crest guidance of soft palate myogenesis
title_fullStr	Runx2-Twist1 interaction coordinates cranial neural crest guidance of soft palate myogenesis
title_full_unstemmed	Runx2-Twist1 interaction coordinates cranial neural crest guidance of soft palate myogenesis
title_sort	runx2-twist1 interaction coordinates cranial neural crest guidance of soft palate myogenesis
publisher	eLife Sciences Publications Ltd
series	eLife
issn	2050-084X
publishDate	2021-01-01
description	Cranial neural crest (CNC) cells give rise to bone, cartilage, tendons, and ligaments of the vertebrate craniofacial musculoskeletal complex, as well as regulate mesoderm-derived craniofacial muscle development through cell-cell interactions. Using the mouse soft palate as a model, we performed an unbiased single-cell RNA-seq analysis to investigate the heterogeneity and lineage commitment of CNC derivatives during craniofacial muscle development. We show that Runx2, a known osteogenic regulator, is expressed in the CNC-derived perimysial and progenitor populations. Loss of Runx2 in CNC-derivatives results in reduced expression of perimysial markers (Aldh1a2 and Hic1) as well as soft palate muscle defects in Osr2-Cre;Runx2fl/fl mice. We further reveal that Runx2 maintains perimysial marker expression through suppressing Twist1, and that myogenesis is restored in Osr2-Cre;Runx2fl/fl;Twist1fl/+ mice. Collectively, our findings highlight the roles of Runx2, Twist1, and their interaction in regulating the fate of CNC-derived cells as they guide craniofacial muscle development through cell-cell interactions.
topic	cleft palate Runx2 cell-cell interaction muscle development cranial neural crest cells
url	https://elifesciences.org/articles/62387
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Runx2-Twist1 interaction coordinates cranial neural crest guidance of soft palate myogenesis

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