SLC37A4, gene responsible for glycogen storage disease type 1b, regulates gingival epithelial barrier function via JAM1 expression

Tanigaki, Keita; Matsumura, Risako; Sasaki, Naoko; Kato, Yuta; Tamamori, Tsukasa; Yamaga, Shunsuke; Nakamura, Eriko; Sakanaka, Akito; Kuboniwa, Masae; Matsusaki, Michiya; Amano, Atsuo; Takeuchi, Hiroki

doi:https://doi.org/10.1038/s41598-024-75524-9

SLC37A4, gene responsible for glycogen storage disease type 1b, regulates gingival epithelial barrier function via JAM1 expression

Tanigaki, Keita; Matsumura, Risako; Sasaki, Naoko et al.

Scientific Reports, 2024, 14, 24797

アクセス数:1,242件（2025-07-26 20:25 集計）

固定URL: https://hdl.handle.net/11094/98556

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タイトル	SLC37A4, gene responsible for glycogen storage disease type 1b, regulates gingival epithelial barrier function via JAM1 expression
著者	NRID 1000071004652 1000071004652 著者名 Tanigaki, Keita
	著者名 Matsumura, Risako
	NRID 1000010388762 1000010388762 researchmap permalink sasaki_n sasaki_n 著者名 Sasaki, Naoko
	著者名 Kato, Yuta
	著者名 Tamamori, Tsukasa
	NRID 1000050964863 1000050964863 著者名 Yamaga, Shunsuke
	NRID 1000000755069 1000000755069 researchmap permalink nakamuraeriko nakamuraeriko Scopus Author ID 56025920800 56025920800 著者名 Nakamura, Eriko
	NRID 1000090815557 1000090815557 researchmap permalink sknk1022 sknk1022 Scopus Author ID 56814868300 56814868300 ORCID 0000-0001-9733-6886 0000-0001-9733-6886 著者名 Sakanaka, Akito
	NRID 1000000303983 1000000303983 ORCID 0000-0002-3332-2431 0000-0002-3332-2431 researchmap permalink MasaeKuboniwa_URL MasaeKuboniwa_URL Scopus Author ID 56089471500 56089471500 著者名 Kuboniwa, Masae
	NRID 1000000419467 1000000419467 researchmap permalink read0093431 read0093431 Scopus Author ID 6701561701 6701561701 著者名 Matsusaki, Michiya
	NRID 1000050193024 1000050193024 researchmap permalink read0014208 read0014208 Scopus Author ID 7101649822 7101649822 ORCID 0000-0002-5158-147X 0000-0002-5158-147X 著者名 Amano, Atsuo
	NRID 1000040572186 1000040572186 researchmap permalink takeuchih takeuchih Scopus Author ID 55007392300 55007392300 著者名 Takeuchi, Hiroki
内容	Tanigaki K., Matsumura R., Sasaki N., et al. SLC37A4, gene responsible for glycogen storage disease type 1b, regulates gingival epithelial barrier function via JAM1 expression. Scientific Reports 14, 24797 (2024); https://doi.org/10.1038/s41598-024-75524-9.
抄録	Solute carrier family 37 member 4 (SLC37A4) is known to regulate glucose-6-phosphate transport from cytoplasm to the lumen of the endoplasmic reticulum, which serves to maintain glucose homeostasis. Glycogen storage disease type 1b (GSD1b) is caused by a mutation of SLC37A4, leading to a glycogenolysis defect. Although GSD1b cases are known to be complicated by periodontitis, the etiological molecular basis remains unclear. The present study investigated the effects of SLC37A4 on gingival barrier function. Examinations of immortalized human gingival epithelial (IHGE) cells showed SLC37A4 localized in the endoplasmic reticulum. SLC37A4 knockout decreased expression of JAM1, a tight junction-related protein, in IHGE cells. Using in silico analysis to investigate potential transcription factor binding sites, H6 family homeobox 3 (HMX3) was shown to be related to JAM1 expression. In HMX3-knockdown IHGE cells, JAM1 expression was markedly suppressed. Furthermore, HMX3 was scarcely detected in SLC37A4-knockout cells, while HMX3 overexpression restored JAM1 expression in those cells. Finally, using a three-dimensional multilayered gingival epithelial tissue model, knockout of SLC37A4 was also found to increase permeability to lipopolysaccharide and peptidoglycan, which was dependent on JAM1 expression. Specific downregulation of HMX3 by SLC37A4 and the consequent decrease in JAM1 expression provides findings indicating a molecular basis for the reduction in barrier function of gingival epithelial tissues in GSD1b cases.
出版者	Nature Research
収録物名	Scientific Reports
巻(号)	14
ページ	24797
刊行年月	2024-10-22
言語	英語
ハンドルURL	https://hdl.handle.net/11094/98556
eISSN	20452322
関連情報 (isIdenticalTo)	DOI (出版社版) https://doi.org/10.1038/s41598-024-75524-9
アクセス権	open access
権利情報	This article is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 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	Scientific Reports. 2024, 14, p. 24797
DC.title	SLC37A4, gene responsible for glycogen storage disease type 1b, regulates gingival epithelial barrier function via JAM1 expression
DC.creator	Tanigaki, Keita
	Matsumura, Risako
	Sasaki, Naoko
	Kato, Yuta
	Tamamori, Tsukasa
	Yamaga, Shunsuke
	Nakamura, Eriko
	Sakanaka, Akito
	Kuboniwa, Masae
	Matsusaki, Michiya
	Amano, Atsuo
	Takeuchi, Hiroki
DC.publisher	Nature Research
DC.language' scheme='DCTERMS.RFC1766	英語
DCTERMS.issued' scheme='DCTERMS.W3CDTF	2024-10-22
DC.identifier	https://doi.org/10.1038/s41598-024-75524-9
DC.description	Tanigaki K., Matsumura R., Sasaki N., et al. SLC37A4, gene responsible for glycogen storage disease type 1b, regulates gingival epithelial barrier function via JAM1 expression. Scientific Reports 14, 24797 (2024); https://doi.org/10.1038/s41598-024-75524-9.
DCTERMS.abstract	Solute carrier family 37 member 4 (SLC37A4) is known to regulate glucose-6-phosphate transport from cytoplasm to the lumen of the endoplasmic reticulum, which serves to maintain glucose homeostasis. Glycogen storage disease type 1b (GSD1b) is caused by a mutation of SLC37A4, leading to a glycogenolysis defect. Although GSD1b cases are known to be complicated by periodontitis, the etiological molecular basis remains unclear. The present study investigated the effects of SLC37A4 on gingival barrier function. Examinations of immortalized human gingival epithelial (IHGE) cells showed SLC37A4 localized in the endoplasmic reticulum. SLC37A4 knockout decreased expression of JAM1, a tight junction-related protein, in IHGE cells. Using in silico analysis to investigate potential transcription factor binding sites, H6 family homeobox 3 (HMX3) was shown to be related to JAM1 expression. In HMX3-knockdown IHGE cells, JAM1 expression was markedly suppressed. Furthermore, HMX3 was scarcely detected in SLC37A4-knockout cells, while HMX3 overexpression restored JAM1 expression in those cells. Finally, using a three-dimensional multilayered gingival epithelial tissue model, knockout of SLC37A4 was also found to increase permeability to lipopolysaccharide and peptidoglycan, which was dependent on JAM1 expression. Specific downregulation of HMX3 by SLC37A4 and the consequent decrease in JAM1 expression provides findings indicating a molecular basis for the reduction in barrier function of gingival epithelial tissues in GSD1b cases.
DC.rights	This article is licensed under a Creative Commons Attribution-NonCommercial-NoDerivatives 4.0 International License.
DC.format	98556
citation_title	SLC37A4, gene responsible for glycogen storage disease type 1b, regulates gingival epithelial barrier function via JAM1 expression
citation_author	Tanigaki, Keita
	Matsumura, Risako
	Sasaki, Naoko
	Kato, Yuta
	Tamamori, Tsukasa
	Yamaga, Shunsuke
	Nakamura, Eriko
	Sakanaka, Akito
	Kuboniwa, Masae
	Matsusaki, Michiya
	Amano, Atsuo
	Takeuchi, Hiroki
citation_publisher	Nature Research
citation_language	英語
citation_date	2024-10-22
citation_journal_title	Scientific Reports
citation_volume	14
citation_firstpage	24797
citation_doi	https://doi.org/10.1038/s41598-024-75524-9
citation_public_url	https://hdl.handle.net/11094/98556

SLC37A4, gene responsible for glycogen storage disease type 1b, regulates gingival epithelial barrier function via JAM1 expression

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