Clear-cell odontogenic carcinoma with pulmonary metastases resembling pulmonary meningothelial-like nodules

Abstract

Clear-cell odontogenic carcinoma (CCOC) is a rare neoplasm with malignant potential and unknown cytogenetic alterations. We describe the case of a 43-year-old woman who presented with an unusual odontogenic epithelial tumor. Histologically, the tumor was composed of clear-cell areas and exhibited a squamous pattern with little nuclear pleomorphism similar to benign squamous odontogenic tumor. Multiple small pulmonary nodules occurring 3 years after primary surgical treatment histologically closely resembled benign minute pulmonary meningothelial-like nodules (MPMN) with clear-cell features. Comparative genomic hybridization (CGH) and immunohistochemistry, performed as diagnostic adjuncts, revealed in the odontogenic tumor and the pulmonary lesions a very similar pattern of chromosomal aberrations (loss of 9, gains of 14q, 19 and 20 in both, and additional loss of 6 in the odontogenic tumor) and the same pattern of expression (positive for cytokeratin 5, 6, 8, 19 and negative for cytokeratin 18, epithelial membrane antigen, and vimentin), differing from that of MPMN. These findings confirmed the final diagnosis of metastasizing CCOC with partial squamous differentiation, substantiated the unfavorable prognosis of the clear-cell component, and highlighted the diagnostic impact of CGH and immunohistochemistry for classification of these morphologically peculiar pulmonary CCOC metastases.

Clear cell odontogenic carcinoma: report of 7 new cases and systematic review of the current knowledge

“…CKs 7, 8, and 18 were absent or mildly expressed, similar to findings in other investigations. 18,19,25,27,35,40,47,52,55 Other antigens have been reported as negative (vimentin, desmin, enolase, smooth muscle actin, calponin, S-100 protein, human melanoma black-45, a(1)-chymotrypsin, CD10, CD31, CD45, glial fibrillary acidic protein, and chromogranin) or mildly positive in expression (CK13 and EMA). 8,18,19,26,27,31,40,47 The previously metioned immunohistochemical markers can be useful in the differential diagnosis of epithelial-myoepithelial salivary gland tumors, melanocytic lesions, and lymphatic tumors.…”

Progesterone receptor immunoreactivity in minute meningothelioid nodules of the lung

“…The occurrence of meningothelial proliferations outside the skull is well known [2,12]. The low proliferative fraction and the lack of mitotic figures or cellular atypia suggest that these lesions are benign, slow-growing proliferations of arachnoid-like cells, although their reactive of neoplastic nature is still debated.…”

Malignant Odontogenic Tumors: An Update on Selected Tumors

“…Hansen originally described it in 1985 as a benign but aggressive lesion, centrally located in the jaws, under the guise of clear cell odontogenic tumor [20]. Subsequent case reports and small series included additional clinical follow-up that documented the capacity of this neoplasm for local destructive growth with invasion of medullary bone, nerves, lymphatics, as well as regional lymph node and distant metastases (pulmonary, bone) [21][22][23]. The World Health Organization reclassified the tumor in 2005 as an odontogenic carcinoma ( Table 1).…”