Enamel defects in Acp4


Journal

Scientific reports
ISSN: 2045-2322
Titre abrégé: Sci Rep
Pays: England
ID NLM: 101563288

Informations de publication

Date de publication:
01 10 2022
Historique:
received: 01 06 2022
accepted: 16 09 2022
entrez: 1 10 2022
pubmed: 2 10 2022
medline: 5 10 2022
Statut: epublish

Résumé

Human ACP4 (OMIM*606362) encodes a transmembrane protein that belongs to histidine acid phosphatase (ACP) family. Recessive mutations in ACP4 cause non-syndromic hypoplastic amelogenesis imperfecta (AI1J, OMIM#617297). While ACP activity has long been detected in developing teeth, its functions during tooth development and the pathogenesis of ACP4-associated AI remain largely unknown. Here, we characterized 2 AI1J families and identified a novel ACP4 disease-causing mutation: c.774_775del, p.Gly260Aspfs*29. To investigate the role of ACP4 during amelogenesis, we generated and characterized Acp4

Identifiants

pubmed: 36183038
doi: 10.1038/s41598-022-20684-9
pii: 10.1038/s41598-022-20684-9
pmc: PMC9526733
doi:

Substances chimiques

Dental Enamel Proteins 0
Histidine 4QD397987E
ACP4 protein, human EC 3.1.3.2
Acid Phosphatase EC 3.1.3.2

Types de publication

Journal Article Research Support, U.S. Gov't, Non-P.H.S. Research Support, Non-U.S. Gov't Research Support, N.I.H., Extramural

Langues

eng

Sous-ensembles de citation

IM

Pagination

16477

Subventions

Organisme : NIH HHS
ID : DE015846
Pays : United States
Organisme : NIDCR NIH HHS
ID : UH3 DE028849
Pays : United States
Organisme : NIDCR NIH HHS
ID : R01 DE015846
Pays : United States
Organisme : NIH HHS
ID : S10 OD028612
Pays : United States
Organisme : NIDCR NIH HHS
ID : R56 DE015846
Pays : United States

Informations de copyright

© 2022. The Author(s).

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Auteurs

Tian Liang (T)

Department of Biologic and Materials Sciences, University of Michigan School of Dentistry, 1011 N University Ave, Ann Arbor, MI, 48109, USA.

Shih-Kai Wang (SK)

Department of Dentistry, National Taiwan University School of Dentistry, No. 1, Changde St., Zhongzheng Dist., Taipei City, 100, Taiwan.
Department of Pediatric Dentistry, National Taiwan University Children's Hospital, No. 8, Zhongshan S. Rd., Zhongzheng Dist., Taipei City, 100, Taiwan.

Charles Smith (C)

Department of Biologic and Materials Sciences, University of Michigan School of Dentistry, 1011 N University Ave, Ann Arbor, MI, 48109, USA.
Department of Anatomy & Cell Biology, Faculty of Medicine & Health Sciences, McGill University, Montreal, QC, Canada.

Hong Zhang (H)

Department of Biologic and Materials Sciences, University of Michigan School of Dentistry, 1011 N University Ave, Ann Arbor, MI, 48109, USA.

Yuanyuan Hu (Y)

Department of Biologic and Materials Sciences, University of Michigan School of Dentistry, 1011 N University Ave, Ann Arbor, MI, 48109, USA.

Figen Seymen (F)

Department of Pedodontics, Faculty of Dentistry, Altinbas University, 34147, Istanbul, Turkey.

Mine Koruyucu (M)

Department of Pedodontics, Faculty of Dentistry, Istanbul University, 34116, Istanbul, Turkey.

Yelda Kasimoglu (Y)

Department of Pedodontics, Faculty of Dentistry, Istanbul University, 34116, Istanbul, Turkey.

Jung-Wook Kim (JW)

Department of Molecular Genetics & Dental Research Institute, School of Dentistry, Seoul National University, Seoul, 03080, Republic of Korea.
Department of Pediatric Dentistry & Dental Research Institute, School of Dentistry, Seoul National University, Seoul, 03080, Republic of Korea.

Chuhua Zhang (C)

Department of Biologic and Materials Sciences, University of Michigan School of Dentistry, 1011 N University Ave, Ann Arbor, MI, 48109, USA.

Thomas L Saunders (TL)

Division of Molecular, Medicine and Genetics, Department of Internal Medicine, University of Michigan Medical School, Ann Arbor, MI, 48109, USA.

James P Simmer (JP)

Department of Biologic and Materials Sciences, University of Michigan School of Dentistry, 1011 N University Ave, Ann Arbor, MI, 48109, USA. jsimmer@umich.edu.

Jan C-C Hu (JC)

Department of Biologic and Materials Sciences, University of Michigan School of Dentistry, 1011 N University Ave, Ann Arbor, MI, 48109, USA.

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