Skip to content
Case report Open access

Squamous carcinoma arising from the epithelial lining of odontogenic keratocysts: report of two cases and literature review.

Sep 2026 · Oral surgery, oral medicine, oral pathology and oral radiology · 0 citations · 48 references
Medicine

Abstract

This study aimed to report the clinical, radiographic, and histopathological features of two cases of primary intraosseous squamous cell carcinoma (PIOSCC) arising from odontogenic keratocysts (OKCs) and to review the literature. A 35-year-old woman and a 21-year-old man presented with mandibular swelling for 5 days and 4 months. Case 1 showed a multilocular radiolucency, whereas Case 2 exhibited a unilocular radiolucency; both involved the posterior mandible. Incisional biopsy confirmed OKC, followed by decompression/marsupialization. During follow-up, both lesions underwent malignant transformation. Patients received surgery and radiotherapy and remain disease-free. Among 40 PIOSCC cases arising from OKCs, males predominated (60%, n = 24), with a mean age of 44.2 ± 16.3 years. The mandible was the main affected site, and swelling, pain, and paresthesia were the most common findings. In conclusion, PIOSCC arising from OKC may resemble cysts and benign odontogenic tumors. Pain, swelling, and ill-defined radiographic margins are highly suspicious findings.

Read PDF

Similar papers

Review Open access Oct 2026

Odontogenic Keratocysts in the Anterior Midline Region: A Case Series and Literature Review

This case series describes three midline odontogenic keratocysts (OKCs) and reviews the associated literature. All three patients were male; two lesions were maxillary. All lesions were surgically excised, with no recurrence. A search of PubMed, Embase, and Scopus identified 33 reported cases of midline OKC, including...

Isabella Souza Malta, Bruno de Albuquerque Hayasida, A. Anbinder et al. · 0 citations
Open access Sep 2026

Clear Cell Odontogenic Carcinoma of the Mandible Incidentally Detected During Routine Dental Examination: A Case Report With 8-Year Follow-up

Clear cell odontogenic carcinoma (CCOC) is a rare malignant odontogenic epithelial tumor that primarily affects the mandible. Due to its nonspecific clinical and radiographic features, preoperative diagnosis is often difficult. We report an asymptomatic case of CCOC in an elderly woman in her eighties, in whom a mandib...

Keita Kano, Hideki Yoshimatsu · 0 citations
Open access Sep 2026

Ameloblastic Fibro-Odontoma of the Maxilla in a 4-Year-Old Girl: A Rare Paediatric Case

Ameloblastic fibro-odontoma (AFO) is a rare mixed odontogenic lesion of childhood whose biological nature remains debated. Recent World Health Organization (WHO) classifications group most lesions previously diagnosed as AFO within the spectrum of developing odontomas rather than distinct odontogenic neoplasms....

H. Kadiri, M. Kamil, F. Zouaidia et al. · 0 citations
Review Open access Oct 2026

When intact mucosa hides a malignancy: solid-type primary intraosseous carcinoma (PIOC, NOS) of the mandible: a case report and literature review

Primary intraosseous carcinoma, not otherwise specified (PIOC, NOS) is a rare odontogenic carcinoma that arises within the jawbone from odontogenic epithelial remnants, with no initial connection to the oral mucosa. Its non-specific clinical and radiographic features overlap with benign odontogenic and inflammato...

B. Tarakji, Maya Al-Jokhadar, Wafaa Talas et al. · 0 citations
Case report Open access Sep 2026

Beyond the Midline: An Unusual Presentation of Acanthomatous Ameloblastoma in the Anterior Maxilla

Acanthomatous ameloblastoma is an uncommon histopathological variant of ameloblastoma characterised by extensive squamous metaplasia and keratin formation within odontogenic epithelial islands. It predominantly affects adults between 30 and 60 years of age and most commonly occurs in the posterior mandible; involvement...

Vedika Pillai, Shams Ul Nisa, A. Jamdade et al. · 0 citations
Review Open access Oct 2026

A case report of sclerosing odontogenic carcinoma

Sclerosing odontogenic carcinoma (SOC) is a rare and controversial odontogenic tumour due to its limited reported cases and diagnostic challenges. Recognised by the World Health Organization (WHO) in 2017, its malignant potential and classification as a carcinoma remain subjects of debate. We present a unique case of S...

R. S. B. Medawela, I. Davies, J. Pallot et al. · 0 citations

We use cookies to run the site and, with your consent, for analytics and to show ads. See our Cookie Policy.