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According to reports, the occurrence of carcinomas developing in odontogenic cysts ranges from 1 to 2 per 1,000. Squamous cell carcinoma (SCC) arising from an odontogenic keratocyst (OKC) is a rare malignant transformation that typically presents in the jaws. This form of carcinoma is locally aggressive and often associated with a poor prognosis, especially when the diagnosis is delayed.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Case presentation<\/jats:title>\n                    <jats:p>This report discusses a healthy 13-year-old girl with no medical, dental, or surgical background who visited the Oral and Maxillofacial Surgery Department due to mild, dull discomfort and gradually increasing swelling in the right maxilla, originating from the palatal side for the past six months. The surgical procedure was conducted under general anesthesia during which a biopsy was collected from the patient; subsequent processing involved haematoxylin and eosin (H&amp;E) staining and examination under a light microscope. The diagnosis of the lesion was made as an invasive, moderately differentiated, keratinizing SCC arising within an odontogenic keratocyst. The patient underwent a subtotal maxillectomy. An obturator was placed postoperatively to restore function and aesthetics.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusion<\/jats:title>\n                    <jats:p>SCC arising from an OKC is an exceptionally rare and aggressive entity, particularly in pediatric patients. This case highlights the diagnostic challenge posed by seemingly benign cystic lesions and emphasizes the importance of histopathological evaluation of all excised jaw cysts.<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1186\/s12903-025-07562-2","type":"journal-article","created":{"date-parts":[[2026,1,15]],"date-time":"2026-01-15T02:23:37Z","timestamp":1768443817000},"update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":0,"title":["Surgical removal of unexpected oral squamous cell carcinoma arising from a maxillary odontogenic keratocyst in a young female patient: a case report and literature review"],"prefix":"10.1186","volume":"26","author":[{"ORCID":"https:\/\/orcid.org\/0009-0001-2540-084X","authenticated-orcid":false,"given":"Tasneem A.","family":"Amer","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mai M.","family":"Saleh","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mohammed S.","family":"Alalfy","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2026,1,15]]},"reference":[{"key":"7562_CR1","first-page":"316","volume":"7","author":"S Tamgadge","year":"2013","unstructured":"Tamgadge S, Tamgadge A, Modak N, Bhalerao S. 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