Sclerosing odontogenic carcinoma with benign fibro-osseous lesion of the mandible: An extremely rare case report

Abstract

A case of sclerosing odontogenic carcinoma (SOC) admixed with a benign fibro-osseous lesion (BFOL) is reported herein. A 67-year-old male had paresthesia in the mental region. Computed tomography detected an intragnathic mass that was focally expansile with disappearance of cortical bone, and contained admixed radiolucency and radio-opacity. Under the pathological diagnosis as benign fibro-osseous lesion, it was surgically removed by curettage. Microscopic analysis showed that a few parts of the resected materials contained dispersed thin cords and small nests of epithelial cells accompanied by fibrous stroma. Cellular atypia and mitotic figures were not evident. The diagnosis of BFOL with hyperplastic and metaplastic odontogenic epithelia was ultimately made. Eight months after the operation, the lesion recurred and segmental mandibulectomy was carried out. Histologically, the lesion was predominantly occupied by the fibro-osseous component with irregular-shaped foci of epithelial component. The epithelial component exhibited mostly thin cord or small nest patterns and showed definite perineural infiltration. Immunohistochemically, the epithelial cells were positive for p63, cytokeratin (CK) 6 and CK19, and focally positive for CK7 but negative for vimentin. MIB-1 positive nuclei were inconspicuous. To the best of our knowledge, this report is the first case of SOC with BFOL.

Sclerosing odontogenic carcinoma

“…The authors should have given more objective reasoning as to why they believed that this case of odontogenic squamous cell carcinoma with osseous metaplasia was similar to SOC on the basis of such few figures, whose findings were not sufficient enough to support the authors' claims. Furthermore, in the previous report of SOC, 3,6 no definite component of conventional squamous cell carcinoma was observed in the SOC. The authors should have provided strict descriptions on how to distinguish SOC from different odontogenic tumors (e.g.…”

“…Third, there was no description about the immunohistochemical analysis neither in this letter to the editor 1 nor in the previous report of PIOSCC 2 . In order to support their claim that this tumor was SOC, the immunohistochemical results of p63, CK5/6, CK7, CK8/18, CK19, CK20, MIB‐1, S‐100, SMA, etc., which have all been previously demonstrated as useful differential diagnostic markers of SOC, 3,6 should have been presented as images for diagnosis of exclusion.…”

Malignant Odontogenic Tumors: An Update on Selected Tumors

“…As noted by the original authors, additional case and series reports are needed to study the biology of this tumor process. There are now seven reported cases in the literature [41][42][43][44][45]. Six of the seven cases have been associated with perineural infiltration [42].…”

“…In one case the tumor was associated with a benign fibro-osseous lesion and initially treated with curettage. This patient had a recurrence 8 months later comprised of the epithelial neoplasm and underwent resection [45]. The most recent patient was treated with enucleation only and has had no recurrence in 12 months [42].…”

Sclerosing odontogenic carcinoma

“…In the October 2010 issue of the Pathology International , a fairly Japanese group of oral pathologists from Tokyo, Hiroshima and Osaka described a rare case of sclerosing odontogenic carcinoma (SOC) with features of benign fibro‐osseous lesion (BFOL) 1 . In their paper, Irie et al 1 . claimed that previous to their case only three other examples of SOC have been reported 2 .…”