Retinoic Acid Deficiency Underlies the Etiology of Midfacial Defects

Wu, Y.; Kurosaka, H.; Wang, Q.; Inubushi, T.; Nakatsugawa, K.; Kikuchi, M.; Ohara, H.; Tsujimoto, T.; Natsuyama, S.; Shida, Y.; Sandell, L. L.; Trainor, P. A.; Yamashiro, T.

doi:https://doi.org/10.1177/00220345211062049

Retinoic Acid Deficiency Underlies the Etiology of Midfacial Defects

Wu, Y.; Kurosaka, H.; Wang, Q. et al.

Journal of Dental Research, 2022, 101(6), 686-694

アクセス数:128件（2026-02-15 21:48 集計）

固定URL: https://hdl.handle.net/11094/103593

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タイトル	Retinoic Acid Deficiency Underlies the Etiology of Midfacial Defects
著者	著者名 Wu, Y.
	NRID 1000020509369 1000020509369 ORCID 0000-0002-8956-5467 0000-0002-8956-5467 researchmap permalink kurosakahiroshi kurosakahiroshi Scopus Author ID 11839162500 11839162500 著者名 Kurosaka, H.
	著者名 Wang, Q.
	NRID 1000030550941 1000030550941 ORCID 0000-0001-5303-4763 0000-0001-5303-4763 researchmap permalink opathone0141 opathone0141 Scopus Author ID 23097933700 23097933700 著者名 Inubushi, T.
	NRID 1000040848248 1000040848248 researchmap permalink NakatsugawaK NakatsugawaK Scopus Author ID 57201396219 57201396219 著者名 Nakatsugawa, K.
	著者名 Kikuchi, M.
	researchmap permalink o_h o_h Scopus Author ID 57410377000 57410377000 著者名 Ohara, H.
	NRID 1000070984904 1000070984904 Scopus Author ID 57410081900 57410081900 著者名 Tsujimoto, T.
	NRID 1000061026001 1000061026001 Scopus Author ID 57410524100 57410524100 著者名 Natsuyama, S.
	著者名 Shida, Y.
	著者名 Sandell, L. L.
	著者名 Trainor, P. A.
	NRID 1000070294428 1000070294428 ORCID 0000-0002-4419-9643 0000-0002-4419-9643 researchmap permalink read0051793 read0051793 Scopus Author ID 55416765600 55416765600 著者名 Yamashiro, T.
キーワード等	facial cleft
	mouse
	neural crest cells
	Rdh10
	retinoid signaling
	sonic hedgehog
内容	Wu Y., Kurosaka H., Wang Q., et al. Retinoic Acid Deficiency Underlies the Etiology of Midfacial Defects. Journal of Dental Research 101(6), pp. 686-694. Copyright © 2022 © International Association for Dental Research and American Association for Dental, Oral, and Craniofacial Research. DOI: https://doi.org/10.1177/00220345211062049.
抄録	Embryonic craniofacial development depends on the coordinated outgrowth and fusion of multiple facial primordia, which are populated with cranial neural crest cells and covered by the facial ectoderm. Any disturbance in these developmental events, their progenitor tissues, or signaling pathways can result in craniofacial deformities such as orofacial clefts, which are among the most common birth defects in humans. In the present study, we show that Rdh10 loss of function leads to a substantial reduction in retinoic acid (RA) signaling in the developing frontonasal process during early embryogenesis, which results in a variety of craniofacial anomalies, including midfacial cleft and ectopic chondrogenic nodules. Elevated apoptosis and perturbed cell proliferation in postmigratory cranial neural crest cells and a substantial reduction in Alx1 and Alx3 transcription in the developing frontonasal process were associated with midfacial cleft in Rdh10-deficient mice. More important, expanded Shh signaling in the ventral forebrain, as well as partial abrogation of midfacial defects in Rdh10 mutants via inhibition of Hh signaling, indicates that misregulation of Shh signaling underlies the pathogenesis of reduced RA signaling-associated midfacial defects. Taken together, these data illustrate the precise spatiotemporal function of Rdh10 and RA signaling during early embryogenesis and their importance in orchestrating molecular and cellular events essential for normal midfacial development.
出版者	SAGE Publications Inc.
収録物名	Journal of Dental Research
巻(号)	101(6)
ページ	686-694
刊行年月	2022-01-09
言語	英語
ハンドルURL	https://hdl.handle.net/11094/103593
PISSN	00220345
eISSN	15440591
関連情報 (isVersionOf)	DOI (出版社版) https://doi.org/10.1177/00220345211062049
アクセス権	open access
出版タイプ	Accepted Manuscript
カテゴリ	学術雑誌論文 Journal Article

資源タイプ	学術雑誌論文
ローカル資源タイプ	学術雑誌論文
DCMI資源タイプ	text
DCTERMS.bibliographicCitation	Journal of Dental Research. 2022, 101(6), p. 686-694
DC.title	Retinoic Acid Deficiency Underlies the Etiology of Midfacial Defects
DC.creator	Wu, Y.
	Kurosaka, H.
	Wang, Q.
	Inubushi, T.
	Nakatsugawa, K.
	Kikuchi, M.
	Ohara, H.
	Tsujimoto, T.
	Natsuyama, S.
	Shida, Y.
	Sandell, L. L.
	Trainor, P. A.
	Yamashiro, T.
DC.publisher	SAGE Publications Inc.
DC.language' scheme='DCTERMS.RFC1766	英語
DC.type' scheme='DCTERMS.DCMIType	text
DCTERMS.issued' scheme='DCTERMS.W3CDTF	2022-01-09
DC.identifier	https://doi.org/10.1177/00220345211062049
DC.subject	facial cleft
	mouse
	neural crest cells
	Rdh10
	retinoid signaling
	sonic hedgehog
DC.description	Wu Y., Kurosaka H., Wang Q., et al. Retinoic Acid Deficiency Underlies the Etiology of Midfacial Defects. Journal of Dental Research 101(6), pp. 686-694. Copyright © 2022 © International Association for Dental Research and American Association for Dental, Oral, and Craniofacial Research. DOI: https://doi.org/10.1177/00220345211062049.
DCTERMS.abstract	Embryonic craniofacial development depends on the coordinated outgrowth and fusion of multiple facial primordia, which are populated with cranial neural crest cells and covered by the facial ectoderm. Any disturbance in these developmental events, their progenitor tissues, or signaling pathways can result in craniofacial deformities such as orofacial clefts, which are among the most common birth defects in humans. In the present study, we show that Rdh10 loss of function leads to a substantial reduction in retinoic acid (RA) signaling in the developing frontonasal process during early embryogenesis, which results in a variety of craniofacial anomalies, including midfacial cleft and ectopic chondrogenic nodules. Elevated apoptosis and perturbed cell proliferation in postmigratory cranial neural crest cells and a substantial reduction in Alx1 and Alx3 transcription in the developing frontonasal process were associated with midfacial cleft in Rdh10-deficient mice. More important, expanded Shh signaling in the ventral forebrain, as well as partial abrogation of midfacial defects in Rdh10 mutants via inhibition of Hh signaling, indicates that misregulation of Shh signaling underlies the pathogenesis of reduced RA signaling-associated midfacial defects. Taken together, these data illustrate the precise spatiotemporal function of Rdh10 and RA signaling during early embryogenesis and their importance in orchestrating molecular and cellular events essential for normal midfacial development.
DC.format	103593
citation_title	Retinoic Acid Deficiency Underlies the Etiology of Midfacial Defects
citation_author	Wu, Y.
	Kurosaka, H.
	Wang, Q.
	Inubushi, T.
	Nakatsugawa, K.
	Kikuchi, M.
	Ohara, H.
	Tsujimoto, T.
	Natsuyama, S.
	Shida, Y.
	Sandell, L. L.
	Trainor, P. A.
	Yamashiro, T.
citation_publisher	SAGE Publications Inc.
citation_language	英語
citation_date	2022-01-09
citation_journal_title	Journal of Dental Research
citation_volume	101
citation_issue	6
citation_firstpage	686
citation_lastpage	694
citation_doi	https://doi.org/10.1177/00220345211062049
citation_public_url	https://hdl.handle.net/11094/103593

Retinoic Acid Deficiency Underlies the Etiology of Midfacial Defects

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