{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,8]],"date-time":"2026-10-08T21:41:40Z","timestamp":1791495700713,"version":"4.3.4"},"reference-count":35,"publisher":"Wiley","issue":"6","license":[{"start":{"date-parts":[[2024,3,13]],"date-time":"2024-03-13T00:00:00Z","timestamp":1710288000000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/501100004704","name":"National Research Council of Thailand","doi-asserted-by":"publisher","award":["N42A650229"],"award-info":[{"award-number":["N42A650229"]}],"id":[{"id":"10.13039\/501100004704","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100010724","name":"Health Systems Research Institute","doi-asserted-by":"publisher","award":["66\u2010101"],"award-info":[{"award-number":["66\u2010101"]}],"id":[{"id":"10.13039\/501100010724","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100010724","name":"Health Systems Research Institute","doi-asserted-by":"publisher","award":["66\u2010122"],"award-info":[{"award-number":["66\u2010122"]}],"id":[{"id":"10.13039\/501100010724","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100002873","name":"Chulalongkorn University","doi-asserted-by":"publisher","id":[{"id":"10.13039\/501100002873","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Int Endodontic J"],"published-print":{"date-parts":[[2024,6]]},"abstract":"<jats:title>Abstract<\/jats:title><jats:sec><jats:title>Aim<\/jats:title><jats:p>Loss\u2010of\u2010function mutations in <jats:italic>FAM20A<\/jats:italic> result in amelogenesis imperfecta IG (AI1G) or enamel\u2010renal syndrome, characterized by hypoplastic enamel, ectopic calcification, and gingival hyperplasia, with some cases reporting spontaneous tooth infection. Despite previous reports on the consequence of FAM20A reduction in gingival fibroblasts and transcriptome analyses of AI1G pulp tissues, suggesting its involvement in mineralization and infection, its role in deciduous dental pulp cells (DDP) remains unreported. The aim of this study was to evaluate the properties of DDP obtained from an AI1G patient, providing additional insights into the effects of FAM20A on the mineralization of DDP.<\/jats:p><\/jats:sec><jats:sec><jats:title>Methodology<\/jats:title><jats:p>DDP were obtained from a <jats:italic>FAM20A<\/jats:italic>\u2010AI1G patient (mutant cells) and three healthy individuals. Cellular behaviours were examined using flow cytometry, MTT, attachment and spreading, colony formation, and wound healing assays. Osteogenic induction was applied to DDP, followed by alizarin red S staining to assess their osteogenic differentiation. The expression of FAM20A\u2010related genes, osteogenic genes, and inflammatory genes was analysed using real\u2010time PCR, Western blot, and\/or immunolocalization. Additionally, STRING analysis was performed to predict potential protein\u2013protein interaction networks.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>The mutant cells exhibited a significant reduction in FAM20A mRNA and protein levels, as well as proliferation, migration, attachment, and colony formation. However, normal FAM20A subcellular localization was maintained. Additionally, osteogenic\/odontogenic genes, <jats:italic>OSX<\/jats:italic>, <jats:italic>OPN<\/jats:italic>, <jats:italic>RUNX2<\/jats:italic>, <jats:italic>BSP<\/jats:italic>, and <jats:italic>DSPP<\/jats:italic>, were downregulated, along with upregulated <jats:italic>ALP<\/jats:italic>. STRING analysis suggested a potential correlation between FAM20A and these osteogenic genes. After osteogenic induction, the mutant cells demonstrated reduced mineral deposition and dysregulated expression of osteogenic genes. Remarkably, <jats:italic>FAM20A<\/jats:italic>, <jats:italic>FAM20C<\/jats:italic>, <jats:italic>RUNX2<\/jats:italic>, <jats:italic>OPN<\/jats:italic>, and <jats:italic>OSX<\/jats:italic> were significantly upregulated in the mutant cells, whilst <jats:italic>ALP,<\/jats:italic> and <jats:italic>OCN<\/jats:italic> was downregulated. Furthermore, the mutant cells exhibited a significant increase in inflammatory gene expression, that is, <jats:italic>IL\u20101\u03b2<\/jats:italic> and <jats:italic>TGF\u2010\u03b21<\/jats:italic>, whereas <jats:italic>IL\u20106<\/jats:italic> and <jats:italic>NF\u03baB1<\/jats:italic> expression was significantly reduced.<\/jats:p><\/jats:sec><jats:sec><jats:title>Conclusion<\/jats:title><jats:p>The reduction of FAM20A in mutant DDP is associated with various cellular deficiencies, including delayed proliferation, attachment, spreading, and migration as well as altered osteogenic and inflammatory responses. These findings provide novel insights into the biology of FAM20A in dental pulp cells and shed light on the molecular mechanisms underlying AI1G pathology.<\/jats:p><\/jats:sec>","DOI":"10.1111\/iej.14056","type":"journal-article","created":{"date-parts":[[2024,3,13]],"date-time":"2024-03-13T11:30:22Z","timestamp":1710329422000},"page":"745-758","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":18,"title":["In\u2010depth investigation of FAM20A insufficiency effects on deciduous dental pulp cells: Altered behaviours, osteogenic differentiation, and inflammatory gene expression"],"prefix":"10.1111","volume":"57","author":[{"given":"Kanokwan","family":"Sriwattanapong","sequence":"first","affiliation":[{"name":"Center of Excellence in Genomics and Precision Dentistry, Department of Physiology, Faculty of Dentistry Chulalongkorn University  Bangkok 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