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Solid type primary intraosseous squamous cell carcinoma in the maxilla: report of a new case
© Iino et al.; licensee BioMed Central Ltd. 2013
Received: 21 August 2013
Accepted: 18 December 2013
Published: 31 December 2013
Primary intraosseous squamous cell carcinoma is a rare jaw bone tumor defined as squamous cell carcinoma arising within the jaw, having no initial connection with the oral mucosa and developing from residual odontogenic epithelium or from an odontogenic cyst or tumor. Solid type of this tumor arising in the maxilla is an even rarer presentation, because the majorities derive from cystic lesions and are found in the posterior mandible.
A 36-year-old Japanese man was referred to our clinic with a complaint of pain around the anterior maxillary region on the right side. Intraoral examination identified a firm, non-fluctuant mass with no ulceration in the vestibular region of teeth #11-13. Incisional biopsy was performed, leading to histological diagnosis of moderately differentiated squamous cell carcinoma. Contrast-enhanced computed tomography revealed a destructive tumor with no cystic lesion, >50 mm in diameter. There was no evidence of metastatic disease on chest radiography, upper gastric endoscopy or positron emission tomography. Based on these findings, solid type primary intraosseous squamous cell carcinoma was diagnosed. The patient underwent tumor ablative surgery. The surgical defect was reconstructed using a partially double-folded free radial forearm flap and prefabricated denture-based surgical obturator. The postoperative course was quite good. Neither recurrence nor metastasis had been found as of 3 years and 1 month postoperatively.
To the best of our knowledge, the present case represents only the 10th case of solid type primary intraosseous squamous cell carcinoma arising in the maxilla to be reported.
Primary intraosseous squamous cell carcinoma (PIOSCC) is a rare jaw bone tumor defined as a squamous cell carcinoma (SCC) arising within the jaw, having no initial connection with the oral mucosa, and, presumably, developing from residual odontogenic epithelium or from an odontogenic cyst or tumor . PIOSCC is classified into solid type, derived from keratocystic odontogenic tumor, and derived from odontogenic cysts . The majority of PIOSCCs are found in the posterior mandible and derived from a cystic lesion [2–7]. Solid type PIOSCC arising in the maxilla is thus an even rarer presentation [2–7]. We present herein a new case of successfully treated solid type PIOSCC of the anterior maxilla.
The diagnostic criteria for PIOSCC include absence of an initial connection with the overlying mucosa or skin and exclusion of metastasis from a distant primary tumor by physical and radiographic examination during a follow-up period of at least 6 months . As the present case fulfilled all the criteria listed above and contained no cystic component, this tumor represented solid type PIOSCC in the maxilla. This tumor is thought to arise by direct malignant transformation of odontogenic epithelial rests such as remnants of dental lamina, and reduced enamel epithelium surrounding unerupted/impacted teeth . In this case, the teeth #11 and 12 were removed before the first visit. Therefore, some aberrant epithelial tissue including the rests of Malassez is also the possible origin.
In a review of 200 cases reported up to 2010, Nomura et al.  reported only eight cases (4%) of solid type PIOSCC in the maxilla. In 2011, Mohyuddin and Yao  reported a new case of this tumor. Thus, to the best of our knowledge, the present case represents only the 10th case of solid type PIOSCC arising in the maxilla to be reported.
Standard treatment for PIOSCC includes surgery, radiotherapy, or both, and the prognosis is relatively poor [2–7]. All reports strongly suggest that wide resection of the primary tumor with a sufficient safety margin [2–9]. However, no standard indications or protocols for radio- or chemoradiotherapy have been established. Additional study is needed to identify optimal treatment modalities.
Written informed consent was obtained from the patient for publication of this case report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
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