{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,2]],"date-time":"2026-07-02T03:56:25Z","timestamp":1782964585972,"version":"3.54.5"},"reference-count":14,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2026,4,30]],"date-time":"2026-04-30T00:00:00Z","timestamp":1777507200000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0"},{"start":{"date-parts":[[2026,7,2]],"date-time":"2026-07-02T00:00:00Z","timestamp":1782950400000},"content-version":"vor","delay-in-days":63,"URL":"https:\/\/creativecommons.org\/licenses\/by-nc-nd\/4.0"}],"funder":[{"DOI":"10.13039\/100017950","name":"Shanghai Municipal Health Commission","doi-asserted-by":"publisher","award":["ZHYYZXYYD-202511"],"award-info":[{"award-number":["ZHYYZXYYD-202511"]}],"id":[{"id":"10.13039\/100017950","id-type":"DOI","asserted-by":"publisher"}]},{"name":"Three-year Action Plan for Shanghai TCM Development and Inheritance Program","award":["[ZY(2025-2027)-2-2-1]"],"award-info":[{"award-number":["[ZY(2025-2027)-2-2-1]"]}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["BMC Oral Health"],"DOI":"10.1186\/s12903-026-08478-1","type":"journal-article","created":{"date-parts":[[2026,4,30]],"date-time":"2026-04-30T01:37:17Z","timestamp":1777513037000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["A case of Rapp-Hodgkin syndrome featuring prominent oral leukokeratosis linked to a TP63 gene variant"],"prefix":"10.1186","volume":"26","author":[{"given":"Weiai","family":"Gan","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jie","family":"Wei","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Zhengyan","family":"Zhao","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ying","family":"Zhang","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lan","family":"Wu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2026,4,30]]},"reference":[{"issue":"13","key":"8478_CR1","doi-asserted-by":"publisher","first-page":"1396","DOI":"10.1002\/ajmg.a.31271","volume":"140","author":"T Rinne","year":"2006","unstructured":"Rinne T, Hamel B, van Bokhoven H, Brunner HG. Pattern of p63 mutations and their phenotypes\u2013update. Am J Med Genet A. 2006;140(13):1396\u2013406.","journal-title":"Am J Med Genet A"},{"issue":"2","key":"8478_CR2","doi-asserted-by":"publisher","first-page":"50","DOI":"10.1097\/MCD.0000000000000117","volume":"25","author":"FB Brueggemann","year":"2016","unstructured":"Brueggemann FB, Bartsch O. A recurrent TP63 mutation causing EEC3 and Rapp\u2013Hodgkin syndromes. Clin Dysmorphol. 2016;25(2):50\u20133.","journal-title":"Clin Dysmorphol"},{"issue":"5","key":"8478_CR3","doi-asserted-by":"publisher","first-page":"297","DOI":"10.1111\/j.1399-0004.1982.tb01375.x","volume":"21","author":"MC Silengo","year":"1982","unstructured":"Silengo MC, Davi GF, Bianco R, Costa M, DeMarco A, Verona R, Franceschini P. Distinctive hair changes (pili torti) in Rapp-Hodgkin ectodermal dysplasia syndrome. Clin Genet. 1982;21(5):297\u2013300.","journal-title":"Clin Genet"},{"issue":"2","key":"8478_CR4","first-page":"149","volume":"24","author":"CF Salinas","year":"1988","unstructured":"Salinas CF, Montes GM. Rapp-Hodgkin syndrome: observations on ten cases and characteristic hair changes (pili canaliculi). Birth Defects Orig Artic Ser. 1988;24(2):149\u201368.","journal-title":"Birth Defects Orig Artic Ser"},{"issue":"3","key":"8478_CR5","doi-asserted-by":"publisher","first-page":"215","DOI":"10.4103\/idoj.IDOJ_100_16","volume":"8","author":"M Chatterjee","year":"2017","unstructured":"Chatterjee M, Neema S, Mukherjee S. Rapp Hodgkin Syndrome. Indian dermatology online J. 2017;8(3):215\u20136.","journal-title":"Indian dermatology online J"},{"issue":"11","key":"8478_CR6","first-page":"ZD13","volume":"18","author":"A Biswas","year":"2024","unstructured":"Biswas A, Dutta K, Khatua A, Banerjee A, Puttanavar R. Rapp-Hodgkin Syndrome: A Case Report on its Clinical and Dental Manifestations. J Clin Diagn Res. 2024;18(11):ZD13\u20136.","journal-title":"J Clin Diagn Res"},{"issue":"8","key":"8478_CR7","doi-asserted-by":"publisher","first-page":"1862","DOI":"10.1111\/odi.13704","volume":"27","author":"S Warnakulasuriya","year":"2021","unstructured":"Warnakulasuriya S, Kujan O, Aguirre-Urizar JM, Bagan JV, Gonz\u00e1lez-Moles M, Kerr AR, Lodi G, Mello FW, Monteiro L, Ogden GR, et al. Oral potentially malignant disorders: A consensus report from an international seminar on nomenclature and classification, convened by the WHO Collaborating Centre for Oral Cancer. Oral Dis. 2021;27(8):1862\u201380.","journal-title":"Oral Dis"},{"issue":"5","key":"8478_CR8","doi-asserted-by":"publisher","first-page":"669","DOI":"10.1590\/S0365-05962010000500010","volume":"85","author":"AM Canto","year":"2010","unstructured":"Canto AM, M\u00fcller H, Freitas RR, Santos PS. Oral lichen planus (OLP): clinical and complementary diagnosis. An Bras Dermatol. 2010;85(5):669\u201375.","journal-title":"An Bras Dermatol"},{"key":"8478_CR9","doi-asserted-by":"publisher","first-page":"e9","DOI":"10.1017\/erm.2015.7","volume":"17","author":"W Cai","year":"2015","unstructured":"Cai W, Jiang B, Yu F, Yang J, Chen Z, Liu J, Wei R, Zhao S, Wang X, Liu S. Current approaches to the diagnosis and treatment of white sponge nevus. Expert Rev Mol Med. 2015;17:e9.","journal-title":"Expert Rev Mol Med"},{"issue":"4","key":"8478_CR10","first-page":"271","volume":"26","author":"Guideline for the Diagnosis and Clinical Management of Oral Submucous Fibrosis","year":"2023","unstructured":"Guideline for the Diagnosis and Clinical Management of Oral Submucous Fibrosis. Chin J Dent Res. 2023;26(4):271\u201385.","journal-title":"Chin J Dent Res"},{"issue":"3","key":"8478_CR11","doi-asserted-by":"publisher","first-page":"e45","DOI":"10.1016\/j.tripleo.2009.05.013","volume":"108","author":"D Compilato","year":"2009","unstructured":"Compilato D, Amato S, Campisi G. Resurgence of syphilis: a diagnosis based on unusual oral mucosa lesions. Oral Surg Oral Med Oral Pathol Oral Radiol Endod. 2009;108(3):e45\u201349.","journal-title":"Oral Surg Oral Med Oral Pathol Oral Radiol Endod"},{"issue":"3","key":"8478_CR12","doi-asserted-by":"publisher","first-page":"368","DOI":"10.1002\/1097-0215(20000801)87:3<368::AID-IJC9>3.0.CO;2-J","volume":"87","author":"K Nylander","year":"2000","unstructured":"Nylander K, Coates PJ, Hall PA. Characterization of the expression pattern of p63 alpha and delta Np63 alpha in benign and malignant oral epithelial lesions. Int J Cancer. 2000;87(3):368\u201372.","journal-title":"Int J Cancer"},{"issue":"6729","key":"8478_CR13","doi-asserted-by":"publisher","first-page":"708","DOI":"10.1038\/19531","volume":"398","author":"AA Mills","year":"1999","unstructured":"Mills AA, Zheng B, Wang XJ, Vogel H, Roop DR, Bradley A. p63 is a p53 homologue required for limb and epidermal morphogenesis. Nature. 1999;398(6729):708\u201313.","journal-title":"Nature"},{"issue":"4","key":"8478_CR14","doi-asserted-by":"publisher","first-page":"411","DOI":"10.4161\/cc.3.4.793","volume":"3","author":"MI Koster","year":"2004","unstructured":"Koster MI, Roop DR. Transgenic mouse models provide new insights into the role of p63 in epidermal development. Cell Cycle. 2004;3(4):411\u20133.","journal-title":"Cell Cycle"}],"container-title":["BMC Oral Health"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/article\/10.1186\/s12903-026-08478-1","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12903-026-08478-1.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1186\/s12903-026-08478-1.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2026,7,2]],"date-time":"2026-07-02T03:19:22Z","timestamp":1782962362000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1186\/s12903-026-08478-1"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2026,4,30]]},"references-count":14,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2026,12]]}},"alternative-id":["8478"],"URL":"https:\/\/doi.org\/10.1186\/s12903-026-08478-1","relation":{"has-preprint":[{"id-type":"doi","id":"10.21203\/rs.3.rs-8632040\/v1","asserted-by":"object"}]},"ISSN":["1472-6831"],"issn-type":[{"value":"1472-6831","type":"electronic"}],"subject":[],"published":{"date-parts":[[2026,4,30]]},"assertion":[{"value":"18 January 2026","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"23 April 2026","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"30 April 2026","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"The study protocol was approved by the Ethics Committee of Shanghai Ninth People\u2019s Hospital, Shanghai Jiao Tong University School of Medicine (approval number: SH9H-2025-T583-1). Furthermore, written informed consent was obtained from the patient\u2019s legal guardian for the publication of this case report and any accompanying identifiable clinical images.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethics approval and consent to participate"}},{"value":"Written and signed informed consent to publish the medical history, clinical photos, and radiographs in this case report was obtained from the parents\u2019 of the patient.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}},{"value":"The authors declare no competing interests.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing interests"}}],"article-number":"1158"}}