A variant of calcifying epithelial odontogenic tumor with Langerhans cells

Abstract

A variant of calcifying epithelial odontogenic tumor (CEOT) with Langerhans cells is reported. Compared to a typical CEOT, the tumor islands of this case were thin and composed of a small number of polyhedral epithelial cells. Almost no calcification of homogeneous eosinophilic materials was observed. In addition, clear cells which structurally corresponded to Langerhans cell were intermingled in the epithelial islands. These cells stain positively for S-100 protein, lysozome, MT 1, LN-3 and OKT 6 antibodies, but not for keratin antibody. Electronmicroscopic examination revealed the rod-shaped and racket-shaped structures called Birbeck's granules in the cytoplasm of these clear cells. Our observations indicate a variant case of CEOT with Langerhans cells in tumor nests.

Calcifying Epithelial Odontogenic Tumour with Clear Langerhans Cells: A Novel Variant, Report of a Case and Review of the Literature

“…The clear cells were interpreted to be LC. The finding of PAS negative clear cells in the current case report with eccentrically located indented nuclei, which are immunoreactive for S100 protein, CD1a and the LC marker Langerin, confirms the dendritic nature of the clear cells initially described by Asano et al [1]. However, in contrast to Asano's observation, calcifications were a frequent finding in the present case.…”

“…In 1990, Asano et al [1] described a variant of CEOT with LC. The case was reported to be unusual in that no calcifications were identifiable in the histological sections examined.…”

“…In 1993, Takata et al [2] suggested a correlation between paucity/absence of calcifications in CEOT and the presence of LC. In 2005, Wang et al [3] based on the observations of Asano and Takata et al [1,2], concluded that LC rich non-calcifying CEOT is a distinct entity common in the Asian population with a predilection for the anterior maxillary region.…”

“…This association which has been described in a few isolated case reports, has culminated in the adoption of the contradictory term ''non-calcifying variant of calcifying epithelial odontogenic tumour with LC'' [1][2][3][4]. LC rich non-calcifying CEOT has further been suggested to be a distinct entity common in the Asian population with a predilection for the anterior maxillary region [3].…”

“…While the majority of the case reports [3,4], with the exception of one case [1], have focused on the presence of dendritic LC in CEOT, the exact nature of clear LC in CEOT has not been clarified. In this paper, we report a case of peripheral CEOT with clear LC in a 37 year old black female, showcase its unique clinical, histomorphological and immunohistological features and address the past scientific efforts in characterizing LC rich CEOT.…”