{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,29]],"date-time":"2026-10-29T12:37:52Z","timestamp":1793277472900,"version":"4.5.0"},"reference-count":23,"publisher":"SAGE Publications","license":[{"start":{"date-parts":[[2026,3,18]],"date-time":"2026-03-18T00:00:00Z","timestamp":1773792000000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/journals.sagepub.com\/page\/policies\/text-and-data-mining-license"}],"funder":[{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["17K11829"],"award-info":[{"award-number":["17K11829"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["21K10182"],"award-info":[{"award-number":["21K10182"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100001691","name":"Japan Society for the Promotion of Science","doi-asserted-by":"publisher","award":["24K20025"],"award-info":[{"award-number":["24K20025"]}],"id":[{"id":"10.13039\/501100001691","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["journals.sagepub.com"],"crossmark-restriction":true},"short-container-title":["J Dent Res"],"abstract":"<jats:p>\n                    X-inactivation is the process of dosage compensation to balance X-linked gene expression levels between females (XX) and males (XY). One of the 2 X-chromosomes is randomly selected for inactivation in each cell during embryogenesis and remains inactive throughout life. Therefore, females have 2 types of cells: those with paternal X-chromosome activation and those with maternal X-chromosome activation. However, it remains unclear how X-inactivation is involved in palate and tooth development.\n                    <jats:italic toggle=\"yes\">Ofd1<\/jats:italic>\n                    is located on the X-chromosome. We found cleft palate in all heterozygous\n                    <jats:italic toggle=\"yes\">Ofd1<\/jats:italic>\n                    mutant mice (\n                    <jats:italic toggle=\"yes\">Ofd1<\/jats:italic>\n                    <jats:sup>fl\/WT<\/jats:sup>\n                    ;\n                    <jats:italic toggle=\"yes\">Wnt1Cre<\/jats:italic>\n                    [HET]), even though some cells should activate the normal X-chromosome due to random X-inactivation. Cells with\n                    <jats:italic toggle=\"yes\">Ofd1<\/jats:italic>\n                    mutant X chromosome activation (\n                    <jats:italic toggle=\"yes\">Ofd1<\/jats:italic>\n                    mutant cells) accumulated at the tip of the palatal shelves due to cell segregation caused by a lack of EFNB1 expression, resulting in a cleft palate in all\n                    <jats:italic toggle=\"yes\">Ofd1<\/jats:italic>\n                    <jats:sup>fl\/WT<\/jats:sup>\n                    ;\n                    <jats:italic toggle=\"yes\">Wnt1Cre<\/jats:italic>\n                    (HET) mice. Unlike the palate,\n                    <jats:italic toggle=\"yes\">Ofd1<\/jats:italic>\n                    <jats:sup>fl\/WT<\/jats:sup>\n                    ;\n                    <jats:italic toggle=\"yes\">Wnt1Cre<\/jats:italic>\n                    (HET) mice showed multiple tooth phenotypes, including extra and absent incisors, while a normal number of incisors was also found. Accumulation of\n                    <jats:italic toggle=\"yes\">Ofd1<\/jats:italic>\n                    mutant cells also occurred in the tooth mesenchyme, and these clusters of\n                    <jats:italic toggle=\"yes\">Ofd1<\/jats:italic>\n                    mutant cells failed to initiate incisor tooth formation due to a lack of Wnt signaling. In contrast to palate development, the location of\n                    <jats:italic toggle=\"yes\">Ofd1<\/jats:italic>\n                    mutant cell clusters differed between\n                    <jats:italic toggle=\"yes\">Ofd1<\/jats:italic>\n                    <jats:sup>fl\/WT<\/jats:sup>\n                    ;\n                    <jats:italic toggle=\"yes\">Wnt1Cre<\/jats:italic>\n                    (HET) mice, likely leading to diversity of tooth phenotypes. Thus, X-inactivation exhibits different involvement between palate and tooth development.\n                  <\/jats:p>","DOI":"10.1177\/00220345261428392","type":"journal-article","created":{"date-parts":[[2026,3,18]],"date-time":"2026-03-18T08:06:00Z","timestamp":1773821160000},"update-policy":"https:\/\/doi.org\/10.1177\/sage-journals-update-policy","source":"Crossref","is-referenced-by-count":0,"title":["Distinct Involvement of X-Inactivation in Organogenesis"],"prefix":"10.1177","author":[{"given":"M.","family":"Kawasaki","sequence":"first","affiliation":[{"name":"Division of Oral Anatomy, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"K.","family":"Kawasaki","sequence":"additional","affiliation":[{"name":"Division of Oral Anatomy, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan"},{"name":"Center for Advanced Oral Science, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"M.","family":"Fukushima","sequence":"additional","affiliation":[{"name":"Division of Oral Anatomy, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan"},{"name":"Division of Oral and Maxillofacial Surgery, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"F. Tisna","family":"Sari","sequence":"additional","affiliation":[{"name":"Division of Oral Anatomy, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-0630-610X","authenticated-orcid":false,"given":"V.","family":"Utama","sequence":"additional","affiliation":[{"name":"Division of Oral Anatomy, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0009-0004-4076-7481","authenticated-orcid":false,"given":"A.","family":"Kesuma","sequence":"additional","affiliation":[{"name":"Division of Oral Anatomy, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"A.","family":"Fujita","sequence":"additional","affiliation":[{"name":"Division of Oral Orthodontics, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"J.","family":"Nihara","sequence":"additional","affiliation":[{"name":"Division of Oral Orthodontics, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"K.","family":"Ichikawa","sequence":"additional","affiliation":[{"name":"Division of Oral Orthodontics, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"T.","family":"Kudo","sequence":"additional","affiliation":[{"name":"Division of Oral Orthodontics, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"S.","family":"Ghafoor","sequence":"additional","affiliation":[{"name":"Department of Craniofacial Development and Stem Cell Biology, Dental Institute, King\u2019s College London, Guy\u2019s Hospital, London Bridge, London, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-2116-9561","authenticated-orcid":false,"given":"P.T.","family":"Sharpe","sequence":"additional","affiliation":[{"name":"Department of Craniofacial Development and Stem Cell Biology, Dental Institute, King\u2019s College London, Guy\u2019s Hospital, London Bridge, London, UK"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Y.","family":"Fukusumi","sequence":"additional","affiliation":[{"name":"Department of Cell Biology, Kidney Research Center, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"H.","family":"Kawachi","sequence":"additional","affiliation":[{"name":"Department of Cell Biology, Kidney Research Center, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"T.","family":"Maeda","sequence":"additional","affiliation":[{"name":"Center for Advanced Oral Science, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"B.","family":"Franco","sequence":"additional","affiliation":[{"name":"Telethon Institute of Genetics and Medicine (TIGEM) in Naples, Italy"},{"name":"Medical Genetics at the Federico II University in Naples, Italy"},{"name":"School for advanced studies (Scuola Superiore Meridionale, Genomic and Experimental Medicine Program) Naples, Italy"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-3496-0347","authenticated-orcid":false,"given":"A.","family":"Ohazama","sequence":"additional","affiliation":[{"name":"Division of Oral Anatomy, Niigata University Graduate School of Medical and Dental Sciences, Niigata, Japan"},{"name":"Center of Excellence in Genomics and Precision Dentistry, Faculty of Dentistry, Chulalongkorn University, Bangkok, Thailand"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"179","published-online":{"date-parts":[[2026,3,18]]},"reference":[{"key":"e_1_3_3_2_1","doi-asserted-by":"crossref","unstructured":"Bisgrove BW Yost HJ. 2006. 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