Non-calcifying/Langerhans cell-rich variant of calcifying epithelial odontogenic tumor: A case report and review

Abstract

A calcifying epithelial odontogenic tumor (CEOT) is a rare benign epithelial odontogenic tumor. Histologically, CEOTs are commonly composed of polygonal epithelial cells, eosinophilic amyloid deposits, and calcifications. The non-calcifying/Langerhans cell-rich variant, one of the three variants of CEOTs according to the 2022 World Health Organization classification of head and neck tumors, is rare with few cases reported to date. We present an additional case of a non-calcifying/Langerhans cell-rich CEOT. A 76-year-old woman was referred to our department for painless swelling of the mandibular incisor. Computed tomography showed a 33 × 10 × 18 mm well-defined bone resorption lesion without distention, radiopaque spots, or unerupted teeth in the mandible. Tissue biopsy showed that the tumor was composed of atypical cells with oval nuclei and eosinophilic cytoplasm and many amorphous eosinophilic amyloid-like deposits, which led to the diagnosis of CEOT. Marginal mandibulectomy was performed under general anesthesia from the right mandibular first premolar to the left mandibular canine. The tumor was composed of small nests and strands of odontogenic epithelial cells and many amorphous eosinophilic amyloid-like deposits; however, there was no area of calcification in any tissue section. Immunohistochemical examination showed that the tumor epithelial cells were positive for S-100 protein. Based on these histopathological and immunohistochemical findings, the tumor was diagnosed as a non-calcifying/Langerhans cell-rich CEOT. No local recurrence was detected over the 4 years of follow up.