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

Number of Access:2,355（2025-10-24 12:12 Counts）

Identifier to cite or link to this item: https://hdl.handle.net/11094/100702

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Title	Slc26a2-mediated sulfate metabolism is important in tooth development
Creator	NRID 1000050966710 1000050966710 CreatorName Yoshida, Yuka
	NRID 1000030550941 1000030550941 researchmap permalink opathone0141 opathone0141 Scopus Author ID 23097933700 23097933700 ORCID 0000-0001-5303-4763 0000-0001-5303-4763 CreatorName Inubushi, Toshihiro
	CreatorName Yokoyama, Mika
	CreatorName Nag, Priyanka
	NRID 1000050530490 1000050530490 researchmap permalink Sasaki_JI Sasaki_JI Scopus Author ID 36138008500 36138008500 ORCID 0000-0002-0330-0397 0000-0002-0330-0397 CreatorName Sasaki, Jun-Ichi
	NRID 1000020635403 1000020635403 researchmap permalink read00021209 read00021209 Scopus Author ID 36114737500 36114737500 ORCID 0000-0002-4666-4756 0000-0002-4666-4756 CreatorName Oka, Ayaka
	NRID 1000091000751 1000091000751 CreatorName Murotani, Tomoya
	NRID 1000041000714 1000041000714 CreatorName Kani, Renshiro
	NRID 1000040912457 1000040912457 researchmap permalink shiraishiyuki shiraishiyuki CreatorName Shiraishi, Yuki
	NRID 1000020509369 1000020509369 ORCID 0000-0002-8956-5467 0000-0002-8956-5467 researchmap permalink kurosakahiroshi kurosakahiroshi Scopus Author ID 11839162500 11839162500 CreatorName Kurosaka, Hiroshi
	NRID 1000060635845 1000060635845 ORCID 0000-0002-3322-0515 0000-0002-3322-0515 researchmap permalink yoshifumitakahata yoshifumitakahata Scopus Author ID 22837135800 22837135800 CreatorName Takahata, Yoshifumi
	NRID 1000060294112 1000060294112 ORCID 0000-0001-7279-8363 0000-0001-7279-8363 researchmap permalink MolBiology MolBiology Scopus Author ID 7201418320 7201418320 CreatorName Nishimura, Riko
	NRID 1000080243244 1000080243244 ORCID 0000-0003-3655-4809 0000-0003-3655-4809 researchmap permalink read0014210 read0014210 Scopus Author ID 55994423600 55994423600 CreatorName Imazato, Satoshi
	CreatorName Papagerakis, Petros
	NRID 1000070294428 1000070294428 ORCID 0000-0002-4419-9643 0000-0002-4419-9643 researchmap permalink read0051793 read0051793 Scopus Author ID 55416765600 55416765600 CreatorName Yamashiro, Takashi
Subject	Extracellular matrix
	Matrix biology
	Odontoblasts
	SLC26A2
	Sulfate metabolism
	Tooth development
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.
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.
Publisher	Company of Biologists Ltd
Source Title	DMM Disease Models and Mechanisms
Volume (Issue)	17(12)
Page	dmm052107
Date of Issued	2024-12-10
Language	English
Handle URL	https://hdl.handle.net/11094/100702
PISSN	17548403
eISSN	17548411
Relation.isIdenticalTo	DOI (Publisher Version) https://doi.org/10.1242/dmm.052107
Access Rights	open access
CopyRight	This article is licensed under a Creative Commons Attribution 4.0 International License.
oaire:version	Version of Record
Category	学術雑誌論文 Journal Article

Resource Type	journal article
Local Type	Journal Article
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	English
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	English
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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