{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,5,30]],"date-time":"2026-05-30T01:36:24Z","timestamp":1780104984231,"version":"3.54.0"},"reference-count":10,"publisher":"MDPI AG","issue":"1","license":[{"start":{"date-parts":[[2025,12,19]],"date-time":"2025-12-19T00:00:00Z","timestamp":1766102400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":["JCM"],"abstract":"<jats:p>This article explores a rare and diagnostically challenging form of Crohn\u2019s disease, known as Oral Crohn\u2019s Disease (OCD), in which the condition is confined to the oral cavity without gastrointestinal involvement. Additionally, Crohn\u2019s disease is typically associated with digestive manifestations, and oral lesions may occasionally represent the first- or even the sole- signs of the disease, making diagnosis difficult due to their non-specific presentation. We report the case of a 22-year-old presenting woman suffering from chronic painful gingivitis and macrocheilitis, in the absence of gastrointestinal symptoms. Despite multiple topical treatments and an initial non-specific histopathological report, a multidisciplinary case discussion and re-evaluation of biopsies led to the diagnosis of OCD. Comprehensive gastrointestinal assessments revealed no intestinal involvement. Owing to the persistence of symptoms and resistance to topical therapies, the patient was subsequently treated with an anti\u2013TNF\u03b1 (Tumor Necrosis Factor alpha) biologic agent. To contextualize this case, we conducted a literature review and identified six similar cases published between 2000 and 2025. Reported patients presenting with symptoms such as lip swelling, cheilitis, mucosal ulcerations, and gingivitis. Histopathological findings consistently demonstrate non-caseating granulomas and inflammatory cell infiltration. Most cases responded favorably to corticosteroids, while some required systemic or biologic therapy. The article highlights that OCD remains underrecognized due to its variable clinical presentation and absence of gastrointestinal manifestations. It emphasizes the importance of integrating clinical, histological, and exclusion-based diagnostic criteria and advocates for a multidisciplinary approach involving dental surgeons, dermatologists, pathologists, and gastroenterologists. Early recognition and long-term monitoring are essential, as gastrointestinal involvement may develop years after the onset of oral symptoms.<\/jats:p>","DOI":"10.3390\/jcm15010004","type":"journal-article","created":{"date-parts":[[2025,12,19]],"date-time":"2025-12-19T09:29:16Z","timestamp":1766136556000},"page":"4","update-policy":"https:\/\/doi.org\/10.3390\/mdpi_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["When Crohn\u2019s Disease Is Confined to the Mouth: A Diagnostic Challenge\u2014A Case Report and Review of the Literature"],"prefix":"10.3390","volume":"15","author":[{"ORCID":"https:\/\/orcid.org\/0009-0000-3533-6098","authenticated-orcid":false,"given":"Axel","family":"Vattier","sequence":"first","affiliation":[{"name":"Department of Dentistry, Oral Medicine and Oral Surgery, UFR Odontologie, Brest University Hospital, 29200 Brest, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Justine","family":"Chapuis","sequence":"additional","affiliation":[{"name":"Department of Dentistry, Oral Medicine and Oral Surgery, UFR Odontologie, Brest University Hospital, 29200 Brest, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4219-5219","authenticated-orcid":false,"given":"Marie","family":"Orliaguet","sequence":"additional","affiliation":[{"name":"Laboratoire Interaction Epithelium Neurones, LIEN (UR 4685), Department of Dentistry, Oral Medicine and Oral Surgery, UFR Odontologie, Brest University Hospital, University of Brest, 29200 Brest, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-5906-5920","authenticated-orcid":false,"given":"Amelie","family":"Bourhis","sequence":"additional","affiliation":[{"name":"Department of Pathology, Brest University Hospital, 29200 Brest, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Franck","family":"Cholet","sequence":"additional","affiliation":[{"name":"Department of Gastroenterology, Brest University Hospital, 29200 Brest, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8088-7059","authenticated-orcid":false,"given":"Laurent","family":"Misery","sequence":"additional","affiliation":[{"name":"Laboratoire Interaction Epithelium Neurones, LIEN (UR 4685), Department of Dermatology, UFR Medecine, Brest University Hospital, University of Brest, 29200 Brest, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2418-4444","authenticated-orcid":false,"given":"Sylvie","family":"Boisram\u00e9","sequence":"additional","affiliation":[{"name":"Inserm, EFS, UMR1078, GGB, Department of Dentistry, Oral Medicine and Oral Surgery, UFR Odontologie, Brest University Hospital, France University of Brest, 29200 Brest, France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"1968","published-online":{"date-parts":[[2025,12,19]]},"reference":[{"key":"ref_1","first-page":"e39186","article-title":"Rare Oral Crohn\u2019s Disease: A Case Report","volume":"15","author":"Veiga","year":"2023","journal-title":"Cureus"},{"key":"ref_2","doi-asserted-by":"crossref","first-page":"163","DOI":"10.1111\/j.1875-595X.2002.tb00622.x","article-title":"Oral Aspects of Crohn\u2019s Disease","volume":"52","author":"Scheper","year":"2002","journal-title":"Int. Dent. J."},{"key":"ref_3","first-page":"147","article-title":"Cheilitis granulomatosa revealing Crohn\u2019s disease","volume":"30","author":"Jellali","year":"2018","journal-title":"Pan Afr. Med. J."},{"key":"ref_4","doi-asserted-by":"crossref","first-page":"S139","DOI":"10.4103\/0973-029X.141369","article-title":"Oral Crohn\u2019s Disease","volume":"18","author":"Padmavathi","year":"2014","journal-title":"J. Oral Maxillofac. Pathol."},{"key":"ref_5","doi-asserted-by":"crossref","first-page":"678","DOI":"10.1111\/j.1610-0387.2007.06429.x","article-title":"Early Oral Presentation of Crohn\u2019s Disease","volume":"5","author":"William","year":"2007","journal-title":"J. Dtsch. Dermatol. Ges. J. Ger. Soc. Dermatol."},{"key":"ref_6","doi-asserted-by":"crossref","first-page":"S431","DOI":"10.4103\/0975-7406.100322","article-title":"Oral Crohn\u2019s Disease without Intestinal Manifestations","volume":"4","author":"Harikishan","year":"2012","journal-title":"J. Pharm. Bioallied Sci."},{"key":"ref_7","doi-asserted-by":"crossref","first-page":"bcr2015212300","DOI":"10.1136\/bcr-2015-212300","article-title":"Oral Manifestations of Crohn\u2019s Disease","volume":"2015","author":"Antunes","year":"2015","journal-title":"BMJ Case Rep."},{"key":"ref_8","doi-asserted-by":"crossref","first-page":"336","DOI":"10.1007\/s10266-013-0108-3","article-title":"Oral Manifestation of Crohn\u2019s Disease without Concomitant Gastrointestinal Involvement","volume":"102","author":"Salek","year":"2014","journal-title":"Odontology"},{"key":"ref_9","doi-asserted-by":"crossref","first-page":"355","DOI":"10.1111\/jgh.13866","article-title":"Oral and Upper Gastrointestinal Crohn\u2019s Disease","volume":"33","author":"Laube","year":"2018","journal-title":"J. Gastroenterol. Hepatol."},{"key":"ref_10","first-page":"146","article-title":"A Diagnostic Challenge: Chronic Cutaneous Ulcers as an Isolated Clinical Finding in Metastatic Crohn\u2019s Disease","volume":"115","year":"2023","journal-title":"Rev. Esp. Enferm. Dig."}],"container-title":["Journal of Clinical Medicine"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/www.mdpi.com\/2077-0383\/15\/1\/4\/pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,12,19]],"date-time":"2025-12-19T09:39:28Z","timestamp":1766137168000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.mdpi.com\/2077-0383\/15\/1\/4"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,12,19]]},"references-count":10,"journal-issue":{"issue":"1","published-online":{"date-parts":[[2026,1]]}},"alternative-id":["jcm15010004"],"URL":"https:\/\/doi.org\/10.3390\/jcm15010004","relation":{},"ISSN":["2077-0383"],"issn-type":[{"value":"2077-0383","type":"electronic"}],"subject":[],"published":{"date-parts":[[2025,12,19]]}}}