Primary intra-osseous squamous cell carcinoma arising from dentigerous cysts: A systematic review of the published case reports

Objectives: Primary Intraosseous Squamous Cell Carcinoma (PIOSCC) may develop from the dentigerous cyst (DC). The present systematic review aimed at characterizing and analyzing the so-far-published cases of PIOSCC arising from DC. Methods: A comprehensive search was conducted utilizing PubMed, Web of Science, and Google Scholar for all relevant studies using a variety of relevant keywords. All studies that reported PIOSCC arising from DC were eligible for inclusion. The quality of the included studies was assessed using the JBI Critical Appraisal Tools for case reports. Results: Twenty-seven case report studies including 29 patients with DC were analyzed. Approximately 79 % of the patients were males, with a mean age of 54 years. One case involved a child who was just 1.4-year-old. The mandible was the most frequently affected jaw, and associated with impacted lower third molars. Two crucial criteria for diagnosis of PIOSCC originating from DC was the transition from normal to abnormal cyst epithelium, and the absence of cystic ameloblastoma, and they were reported in 28 cases (96.6 %). Other two criteria, exclusion of the presence of primary carcinoma elsewhere, and communication with the oral mucosa, were reported less frequently (6 and 18 cases, respectively). Several studies incorporated additional evaluation methods such as immunohistochemical, CT, MRI, and PET-CT. In terms of treatment, surgery alone was the first choice, followed by radiation therapy. Conclusions: Despite its rarity, PIOSCC can arise from DC. Once DC is suspected, a prompt surgical excision with robust serial microscopic examination of the whole excised lesion should be done.