{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,6,3]],"date-time":"2026-06-03T08:03:05Z","timestamp":1780473785438,"version":"3.54.1"},"reference-count":76,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2026,5,6]],"date-time":"2026-05-06T00:00:00Z","timestamp":1778025600000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/creativecommons.org\/licenses\/by-nc\/4.0\/"},{"start":{"date-parts":[[2026,5,6]],"date-time":"2026-05-06T00:00:00Z","timestamp":1778025600000},"content-version":"tdm","delay-in-days":0,"URL":"http:\/\/doi.wiley.com\/10.1002\/tdm_license_1.1"}],"funder":[{"DOI":"10.13039\/100014865","name":"Fondation Maladies Rares","doi-asserted-by":"publisher","award":["PRIX APICIL (2024)_0522"],"award-info":[{"award-number":["PRIX APICIL (2024)_0522"]}],"id":[{"id":"10.13039\/100014865","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100003768","name":"Universit\u00e9 de Strasbourg","doi-asserted-by":"publisher","award":["2024\u2013220"],"award-info":[{"award-number":["2024\u2013220"]}],"id":[{"id":"10.13039\/501100003768","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Clinical Genetics"],"published-print":{"date-parts":[[2026,7]]},"abstract":"<jats:title>ABSTRACT<\/jats:title>\n                  <jats:p>\n                    <jats:italic>WNT10A<\/jats:italic>\n                    mutations, a major genetic determinant of dental agenesis and ectodermal dysplasia, exert profound effects on craniofacial development. Although classified as rare disorders, these mutations account for more than half of oligodontia cases, reflecting their critical role. The associated phenotypes span a broad clinical spectrum, ranging from isolated tooth agenesis to complex multisystem disorders, including hypodontia, enamel and root defects, palmoplantar keratoderma, alopecia, nail dystrophy, skin xerosis, hypohidrosis, eyelid cysts, and skin cancer, skeletal abnormalities and impaired bone homeostasis, underscoring their systemic impact. This review synthesizes current biological and clinical knowledge by linking phenotypes of\n                    <jats:italic>WNT10A<\/jats:italic>\n                    mutations to disruptions in Wnt\/\u03b2\u2010catenin signaling and its interactions with\n                    <jats:italic>BMP, SMAD, RANK\/RANKL\/OPG<\/jats:italic>\n                    ,\n                    <jats:italic>VEGF<\/jats:italic>\n                    , and other regulatory pathways. Altered pathway activity affects key processes in tooth morphogenesis, osteogenesis, cellular proliferation, and tissue maintenance, offering mechanistic explanations for the heterogeneity and severity of patient presentations. A clearer understanding of these pathways is essential for improving diagnosis and management of\n                    <jats:italic>WNT10A<\/jats:italic>\n                    \u2010associated oligodontia, particularly in treatment planning for growth\u2010dependent interventions, regenerative strategies, and implant rehabilitation. Despite significant progress, critical gaps remain in defining the molecular basis of variable expressivity and genotype\u2013phenotype relationships. Elucidating these mechanisms will be pivotal for advancing precision approaches in dental and craniofacial care.\n                  <\/jats:p>","DOI":"10.1111\/cge.70178","type":"journal-article","created":{"date-parts":[[2026,5,6]],"date-time":"2026-05-06T13:00:03Z","timestamp":1778072403000},"page":"3-14","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Molecular Basis and Clinical Spectrum of\n                    <i>WNT10A<\/i>\n                    \u2010Related Oligodontia"],"prefix":"10.1111","volume":"110","author":[{"ORCID":"https:\/\/orcid.org\/0009-0003-0604-491X","authenticated-orcid":false,"given":"Perennes","family":"Elise","sequence":"first","affiliation":[{"name":"French National Institute of Health and Medical Research (INSERM), UMR 1260, Regenerative Nanomedicine (RNM), FMTS, CRBS  Strasbourg France"},{"name":"Universit\u00e9 de Strasbourg, Facult\u00e9 de Chirurgie Dentaire  Strasbourg France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3059-3559","authenticated-orcid":false,"given":"Benkirane\u2010Jessel","family":"Nadia","sequence":"additional","affiliation":[{"name":"French National Institute of Health and Medical Research (INSERM), UMR 1260, Regenerative Nanomedicine (RNM), FMTS, CRBS  Strasbourg France"},{"name":"Universit\u00e9 de Strasbourg, Facult\u00e9 de Chirurgie Dentaire  Strasbourg France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anton","family":"Nicolas","sequence":"additional","affiliation":[{"name":"French National Institute of Health and Medical Research (INSERM), UMR 1260, Regenerative Nanomedicine (RNM), FMTS, CRBS  Strasbourg France"},{"name":"Universit\u00e9 de Strasbourg, Facult\u00e9 de Pharmacie  Strasbourg France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Vandamme","family":"Thierry","sequence":"additional","affiliation":[{"name":"French National Institute of Health and Medical Research (INSERM), UMR 1260, Regenerative Nanomedicine (RNM), FMTS, CRBS  Strasbourg France"},{"name":"Universit\u00e9 de Strasbourg, Facult\u00e9 de Pharmacie  Strasbourg France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8078-2406","authenticated-orcid":false,"given":"Clauss","family":"Fran\u00e7ois","sequence":"additional","affiliation":[{"name":"French National Institute of Health and Medical Research (INSERM), UMR 1260, Regenerative Nanomedicine (RNM), FMTS, CRBS  Strasbourg France"},{"name":"Universit\u00e9 de Strasbourg, Facult\u00e9 de Chirurgie Dentaire  Strasbourg France"},{"name":"Universit\u00e9 de Strasbourg, UMR 1260, CRBS  Strasbourg France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-8002-5894","authenticated-orcid":false,"given":"Conzatti","family":"Guillaume","sequence":"additional","affiliation":[{"name":"French National Institute of Health and Medical Research (INSERM), UMR 1260, Regenerative Nanomedicine (RNM), FMTS, CRBS  Strasbourg France"},{"name":"Universit\u00e9 de Strasbourg, Facult\u00e9 de Pharmacie  Strasbourg France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2026,5,6]]},"reference":[{"key":"e_1_2_10_2_1","doi-asserted-by":"publisher","DOI":"10.1086\/520064"},{"key":"e_1_2_10_3_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.36520"},{"key":"e_1_2_10_4_1","doi-asserted-by":"publisher","DOI":"10.1111\/bjd.20601"},{"key":"e_1_2_10_5_1","doi-asserted-by":"publisher","DOI":"10.1136\/jmedgenet\u20102012\u2010100750"},{"key":"e_1_2_10_6_1","doi-asserted-by":"publisher","DOI":"10.1007\/978\u20103\u2010540\u201077727\u20104"},{"key":"e_1_2_10_7_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ajhg.2009.06.001"},{"key":"e_1_2_10_8_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bone.2011.08.010"},{"key":"e_1_2_10_9_1","doi-asserted-by":"publisher","DOI":"10.1038\/ncomms15397"},{"key":"e_1_2_10_10_1","doi-asserted-by":"publisher","DOI":"10.3892\/ol.2019.10035"},{"key":"e_1_2_10_11_1","doi-asserted-by":"publisher","DOI":"10.1161\/CIRCRESAHA.110.219840"},{"key":"e_1_2_10_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cellsig.2009.11.021"},{"key":"e_1_2_10_13_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.devcel.2009.06.016"},{"key":"e_1_2_10_14_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12895\u2010016\u20100040\u20107"},{"key":"e_1_2_10_15_1","doi-asserted-by":"publisher","DOI":"10.4103\/idoj.IDOJ_26_18"},{"key":"e_1_2_10_16_1","doi-asserted-by":"publisher","DOI":"10.1111\/j.1440\u20100960.2011.00788.x"},{"key":"e_1_2_10_17_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.36243"},{"key":"e_1_2_10_18_1","doi-asserted-by":"publisher","DOI":"10.1002\/ajmg.a.32869"},{"key":"e_1_2_10_19_1","doi-asserted-by":"publisher","DOI":"10.7150\/ijms.26997"},{"key":"e_1_2_10_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bone.2018.10.009"},{"key":"e_1_2_10_21_1","doi-asserted-by":"publisher","DOI":"10.7150\/ijms.71127"},{"key":"e_1_2_10_22_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0195156"},{"key":"e_1_2_10_23_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ydbio.2009.07.015"},{"key":"e_1_2_10_24_1","doi-asserted-by":"publisher","DOI":"10.1101\/gad.1035602"},{"key":"e_1_2_10_25_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M113.462473"},{"key":"e_1_2_10_26_1","doi-asserted-by":"publisher","DOI":"10.1016\/0003\u20109969(95)00047\u2010s"},{"key":"e_1_2_10_27_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0092-8674(05)80083-2"},{"key":"e_1_2_10_28_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.081927"},{"key":"e_1_2_10_29_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.122.10.3035"},{"key":"e_1_2_10_30_1","doi-asserted-by":"publisher","DOI":"10.1038\/ejhg.2016.117"},{"key":"e_1_2_10_31_1","doi-asserted-by":"publisher","DOI":"10.33549\/physiolres.930766"},{"issue":"7","key":"e_1_2_10_32_1","first-page":"1537","article-title":"Murine Wnt10a and Wnt10b: Cloning and Expression in Developing Limbs, Face and Skin of Embryos and in Adults","volume":"13","author":"Wang J.","year":"1996","journal-title":"Oncogene"},{"key":"e_1_2_10_33_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41467\u2010018\u201007459\u20105"},{"key":"e_1_2_10_34_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M110.137273"},{"key":"e_1_2_10_35_1","doi-asserted-by":"publisher","DOI":"10.3390\/jcm5040041"},{"key":"e_1_2_10_36_1","doi-asserted-by":"publisher","DOI":"10.1371\/journal.pone.0103240"},{"key":"e_1_2_10_37_1","doi-asserted-by":"publisher","DOI":"10.12998\/wjcc.v11.i34.8094"},{"key":"e_1_2_10_38_1","doi-asserted-by":"publisher","DOI":"10.1159\/000447896"},{"key":"e_1_2_10_39_1","doi-asserted-by":"publisher","DOI":"10.1242\/dev.109231"},{"key":"e_1_2_10_40_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.ophtha.2017.05.031"},{"key":"e_1_2_10_41_1","doi-asserted-by":"publisher","DOI":"10.1146\/annurev\u2010physiol\u2010022516\u2010034202"},{"key":"e_1_2_10_42_1","doi-asserted-by":"publisher","DOI":"10.1038\/ng1932"},{"key":"e_1_2_10_43_1","doi-asserted-by":"publisher","DOI":"10.1177\/0022034510363373"},{"key":"e_1_2_10_44_1","doi-asserted-by":"publisher","DOI":"10.1101\/gad.1457106"},{"key":"e_1_2_10_45_1","doi-asserted-by":"publisher","DOI":"10.1038\/nature12214"},{"key":"e_1_2_10_46_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jid.2016.03.018"},{"key":"e_1_2_10_47_1","doi-asserted-by":"publisher","DOI":"10.1016\/s0925\u20104773(01)00452\u2010x"},{"key":"e_1_2_10_48_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.mpsur.2022.10.011"},{"key":"e_1_2_10_49_1","doi-asserted-by":"publisher","DOI":"10.1111\/scd.12228"},{"key":"e_1_2_10_50_1","doi-asserted-by":"publisher","DOI":"10.1172\/JCI39104"},{"key":"e_1_2_10_51_1","doi-asserted-by":"publisher","DOI":"10.1038\/onc.2011.373"},{"key":"e_1_2_10_52_1","doi-asserted-by":"publisher","DOI":"10.1093\/carcin\/bgv025"},{"key":"e_1_2_10_53_1","doi-asserted-by":"publisher","DOI":"10.1093\/carcin\/bgl161"},{"key":"e_1_2_10_54_1","doi-asserted-by":"publisher","DOI":"10.1111\/cge.12972"},{"key":"e_1_2_10_55_1","doi-asserted-by":"publisher","DOI":"10.1128\/MCB.01428\u201009"},{"key":"e_1_2_10_56_1","doi-asserted-by":"publisher","DOI":"10.1073\/pnas.1300532110"},{"key":"e_1_2_10_57_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M413274200"},{"key":"e_1_2_10_58_1","doi-asserted-by":"publisher","DOI":"10.1369\/0022155411432010"},{"key":"e_1_2_10_59_1","doi-asserted-by":"publisher","DOI":"10.1002\/bies.201200061"},{"key":"e_1_2_10_60_1","doi-asserted-by":"publisher","DOI":"10.1016\/S0006\u2010291X(02)02951\u20100"},{"key":"e_1_2_10_61_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M500608200"},{"key":"e_1_2_10_62_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M111.241505"},{"key":"e_1_2_10_63_1","doi-asserted-by":"publisher","DOI":"10.1038\/s41422\u2010023\u201000918\u20109"},{"key":"e_1_2_10_64_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.diff.2010.05.002"},{"key":"e_1_2_10_65_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bone.2009.03.656"},{"key":"e_1_2_10_66_1","doi-asserted-by":"publisher","DOI":"10.1038\/boneres.2016.9"},{"key":"e_1_2_10_67_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1016\u20108478(23)17408\u20105"},{"key":"e_1_2_10_68_1","doi-asserted-by":"publisher","DOI":"10.1139\/bcb\u20102015\u20100018"},{"key":"e_1_2_10_69_1","doi-asserted-by":"publisher","DOI":"10.1186\/s13018\u2010022\u201003230\u20101"},{"key":"e_1_2_10_70_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.bone.2006.09.022"},{"key":"e_1_2_10_71_1","doi-asserted-by":"publisher","DOI":"10.1002\/jcp.24734"},{"key":"e_1_2_10_72_1","doi-asserted-by":"publisher","DOI":"10.1152\/ajpcell.00278.2004"},{"key":"e_1_2_10_73_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M112.437921"},{"key":"e_1_2_10_74_1","doi-asserted-by":"publisher","DOI":"10.1074\/jbc.M111.256768"},{"key":"e_1_2_10_75_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.cellsig.2014.07.018"},{"key":"e_1_2_10_76_1","doi-asserted-by":"publisher","DOI":"10.1007\/s001980070028"},{"key":"e_1_2_10_77_1","doi-asserted-by":"publisher","DOI":"10.1186\/ar2165"}],"container-title":["Clinical Genetics"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/cge.70178","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/full-xml\/10.1111\/cge.70178","content-type":"application\/xml","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1111\/cge.70178","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,6,3]],"date-time":"2026-06-03T07:19:27Z","timestamp":1780471167000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1111\/cge.70178"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,5,6]]},"references-count":76,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2026,7]]}},"alternative-id":["10.1111\/cge.70178"],"URL":"https:\/\/doi.org\/10.1111\/cge.70178","archive":["Portico"],"relation":{},"ISSN":["0009-9163","1399-0004"],"issn-type":[{"value":"0009-9163","type":"print"},{"value":"1399-0004","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,5,6]]},"assertion":[{"value":"2026-03-13","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-04-15","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2026-05-06","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}