Slc26a2-mediated sulfate metabolism is important in tooth development

Yoshida, Yuka; Inubushi, Toshihiro; Yokoyama, Mika; Nag, Priyanka; Sasaki, Jun-Ichi; Oka, Ayaka; Murotani, Tomoya; Kani, Renshiro; Shiraishi, Yuki; Kurosaka, Hiroshi; Takahata, Yoshifumi; Nishimura, Riko; Imazato, Satoshi; Papagerakis, Petros; Yamashiro, Takashi

doi:https://doi.org/10.1242/dmm.052107

Slc26a2-mediated sulfate metabolism is important in tooth development

Yoshida, Yuka; Inubushi, Toshihiro; Yokoyama, Mika et al.

DMM Disease Models and Mechanisms, 2024, 17(12), dmm052107

アクセス数:24件（2025-04-22 07:22 集計）

固定URL: https://hdl.handle.net/11094/100702

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タイトル	Slc26a2-mediated sulfate metabolism is important in tooth development
著者	NRID 1000050966710 1000050966710 著者名 Yoshida, Yuka
	NRID 1000030550941 1000030550941 researchmap permalink opathone0141 opathone0141 Scopus Author ID 23097933700 23097933700 ORCID 0000-0001-5303-4763 0000-0001-5303-4763 著者名 Inubushi, Toshihiro
	著者名 Yokoyama, Mika
	著者名 Nag, Priyanka
	NRID 1000050530490 1000050530490 researchmap permalink Sasaki_JI Sasaki_JI Scopus Author ID 36138008500 36138008500 著者名 Sasaki, Jun-Ichi
	NRID 1000020635403 1000020635403 researchmap permalink read00021209 read00021209 Scopus Author ID 36114737500 36114737500 著者名 Oka, Ayaka
	NRID 1000091000751 1000091000751 著者名 Murotani, Tomoya
	NRID 1000041000714 1000041000714 著者名 Kani, Renshiro
	NRID 1000040912457 1000040912457 researchmap permalink shiraishiyuki shiraishiyuki 著者名 Shiraishi, Yuki
	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, Hiroshi
	NRID 1000060635845 1000060635845 ORCID 0000-0002-3322-0515 0000-0002-3322-0515 researchmap permalink yoshifumitakahata yoshifumitakahata Scopus Author ID 22837135800 22837135800 著者名 Takahata, Yoshifumi
	NRID 1000060294112 1000060294112 ORCID 0000-0001-7279-8363 0000-0001-7279-8363 researchmap permalink MolBiology MolBiology Scopus Author ID 7201418320 7201418320 著者名 Nishimura, Riko
	NRID 1000080243244 1000080243244 ORCID 0000-0003-3655-4809 0000-0003-3655-4809 researchmap permalink read0014210 read0014210 Scopus Author ID 55994423600 55994423600 著者名 Imazato, Satoshi
	著者名 Papagerakis, Petros
	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, Takashi
キーワード等	Extracellular matrix
	Matrix biology
	Odontoblasts
	SLC26A2
	Sulfate metabolism
	Tooth development
内容	Yoshida Y., Inubushi T., Yokoyama M., et al. Slc26a2-mediated sulfate metabolism is important in tooth development. DMM Disease Models and Mechanisms 17, dmm052107 (2024); https://doi.org/10.1242/dmm.052107.
抄録	The sulfate transporter gene SLC26A2 is crucial for skeletal formation, as evidenced by its role in diastrophic dysplasia, a type of skeletal dysplasia in humans. Although SLC26A2-related chondrodysplasia also affects craniofacial and tooth development, its specific role in these processes remains unclear. In this study, we explored the pivotal roles of SLC26A2-mediated sulfate metabolism during tooth development. We found that Slc26a2 was predominantly expressed in dental tissues, including odontoblasts and ameloblasts. Slc26a2 knockout (Slc26a2-KO-Δexon2) mice exhibited distinct craniofacial abnormalities, such as a retrognathic upper jaw, small upper incisors and upper molar hypoplasia. These mice also showed flattened odontoblasts and loss of nuclear polarity in upper incisors and molars, with significant reductions in odontoblast differentiation markers Dspp and Dmp1. Ex vivo and in vitro studies further revealed dentin matrix hypoplasia, tooth root shortening and downregulation of Wnt signaling in Slc26a2-deficient cells. These findings highlight the crucial role of SLC26A2-mediated sulfate metabolism in tooth development and offer insights into the mechanisms underlying dental abnormalities in patients with SLC26A2-related chondrodysplasias.
出版者	Company of Biologists Ltd
収録物名	DMM Disease Models and Mechanisms
巻(号)	17(12)
ページ	dmm052107
刊行年月	2024-12-10
言語	英語
ハンドルURL	https://hdl.handle.net/11094/100702
PISSN	17548403
eISSN	17548411
関連情報 (isIdenticalTo)	DOI (出版社版) https://doi.org/10.1242/dmm.052107
アクセス権	open access
権利情報	This article is licensed under a Creative Commons Attribution 4.0 International License.
出版タイプ	Version of Record
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
DCTERMS.bibliographicCitation	DMM Disease Models and Mechanisms. 2024, 17(12), p. dmm052107
DC.title	Slc26a2-mediated sulfate metabolism is important in tooth development
DC.creator	Yoshida, Yuka
	Inubushi, Toshihiro
	Yokoyama, Mika
	Nag, Priyanka
	Sasaki, Jun-Ichi
	Oka, Ayaka
	Murotani, Tomoya
	Kani, Renshiro
	Shiraishi, Yuki
	Kurosaka, Hiroshi
	Takahata, Yoshifumi
	Nishimura, Riko
	Imazato, Satoshi
	Papagerakis, Petros
	Yamashiro, Takashi
DC.publisher	Company of Biologists Ltd
DC.language' scheme='DCTERMS.RFC1766	英語
DCTERMS.issued' scheme='DCTERMS.W3CDTF	2024-12-10
DC.identifier	https://doi.org/10.1242/dmm.052107
DC.subject	Extracellular matrix
	Matrix biology
	Odontoblasts
	SLC26A2
	Sulfate metabolism
	Tooth development
DC.description	Yoshida Y., Inubushi T., Yokoyama M., et al. Slc26a2-mediated sulfate metabolism is important in tooth development. DMM Disease Models and Mechanisms 17, dmm052107 (2024); https://doi.org/10.1242/dmm.052107.
DCTERMS.abstract	The sulfate transporter gene SLC26A2 is crucial for skeletal formation, as evidenced by its role in diastrophic dysplasia, a type of skeletal dysplasia in humans. Although SLC26A2-related chondrodysplasia also affects craniofacial and tooth development, its specific role in these processes remains unclear. In this study, we explored the pivotal roles of SLC26A2-mediated sulfate metabolism during tooth development. We found that Slc26a2 was predominantly expressed in dental tissues, including odontoblasts and ameloblasts. Slc26a2 knockout (Slc26a2-KO-Δexon2) mice exhibited distinct craniofacial abnormalities, such as a retrognathic upper jaw, small upper incisors and upper molar hypoplasia. These mice also showed flattened odontoblasts and loss of nuclear polarity in upper incisors and molars, with significant reductions in odontoblast differentiation markers Dspp and Dmp1. Ex vivo and in vitro studies further revealed dentin matrix hypoplasia, tooth root shortening and downregulation of Wnt signaling in Slc26a2-deficient cells. These findings highlight the crucial role of SLC26A2-mediated sulfate metabolism in tooth development and offer insights into the mechanisms underlying dental abnormalities in patients with SLC26A2-related chondrodysplasias.
DC.rights	This article is licensed under a Creative Commons Attribution 4.0 International License.
DC.format	100702
citation_title	Slc26a2-mediated sulfate metabolism is important in tooth development
citation_author	Yoshida, Yuka
	Inubushi, Toshihiro
	Yokoyama, Mika
	Nag, Priyanka
	Sasaki, Jun-Ichi
	Oka, Ayaka
	Murotani, Tomoya
	Kani, Renshiro
	Shiraishi, Yuki
	Kurosaka, Hiroshi
	Takahata, Yoshifumi
	Nishimura, Riko
	Imazato, Satoshi
	Papagerakis, Petros
	Yamashiro, Takashi
citation_publisher	Company of Biologists Ltd
citation_language	英語
citation_date	2024-12-10
citation_journal_title	DMM Disease Models and Mechanisms
citation_volume	17
citation_issue	12
citation_firstpage	dmm052107
citation_doi	https://doi.org/10.1242/dmm.052107
citation_public_url	https://hdl.handle.net/11094/100702

Slc26a2-mediated sulfate metabolism is important in tooth development

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