PRIMARY XANTHOMA OF THE MANDIBLE: A CASE REPORT OF A RARE ENTITY

Autor: Sherry Timmons, Juan Pablo Castro Cuellar, Shahidul Ahsan, N. Handoo, Scott M. Steward-Tharp, Felipe Nor, S. Anamali
Rok vydání: 2019
Předmět:
Zdroj: Oral Surgery, Oral Medicine, Oral Pathology and Oral Radiology. 128:e69
ISSN: 2212-4403
DOI: 10.1016/j.oooo.2019.02.166
Popis: Objective Central xanthoma of the jaw is an extraordinarily rare entity with less than 30 cases reported in the English literature so far. This benign lesion is often associated with endocrine and metabolic diseases (e.g. hyperlipidemia, diabetes mellitus, etc.). When those conditions are ruled out, primary xanthoma of the jaw is the appropriate diagnostic term. Adult males are most commonly affected, and the lesion is most frequently reported in the mandible. The classic microscopic features include the presence of histiocytic-like cells with foamy cytoplasm that stain positive by immunohistochemistry for CD68, but are negative for S100 and CD1a. Due to the microscopic similarities of histiocytic-like cells in H/E slides, Erdheim-Chester disease and Rosai-Dorfman disease are two systemic conditions that should be considered in the work-up. Findings We present a case of a 15 years-old male patient with multiple, ill-defined, non-corticated, radiolucent entities in the left ramus of the mandible. The lesions appear to be coalescing in some views. There is no evidence of bucco-lingual expansion and cortical destruction of bone. An excisional biopsy is performed. Microscopic examination reveals mixed soft and hard tissues. The hard tissue is composed of reactive vital bone. Sheets of foamy cells with dark, centrally placed vesiculated nuclei, prominent nucleoli and well-defined cytoplasmic membrane are noted between the bony trabeculae. In some areas, epithelioid cells with more amphophilic cytoplasm and less distinct cytoplasmic membrane are identified. Chronic inflammatory infiltrate with extravasated erythrocytes within the background of the connective tissue is also observed. Conclusion The appropriate diagnosis of central xanthoma of the jaw requires the work-up for systemic diseases in association with immunohistochemical profile. The recommended treatment is excision and curettage, which has been associated with excellent prognosis and extremely low recurrence rate.
Databáze: OpenAIRE
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