Three Cases of Central Xanthoma of the Jaws with Review of Literature

Abstract

<h3>Background</h3> Extragnathic xanthomas are rare benign tumors associated with excess lipid production. Its gnathic counterpart was first reported in 1964. Only 45 cases of central xanthoma of jaw (CXJ) have been reported in the English literature. The etiopathogenesis of CXJ is highly suggestive of a reactive process or a metabolic condition. <h3>Materials</h3> Three cases of CXJ were identified from the UF Oral Pathology Biopsy archives. Clinical, radiographic and histopathologic features of all the cases were retrospectively analyzed. Immunohistochemical (IHC) stains for S100 and CD68 were performed. <h3>Case Series</h3> All cases of CXJ involved younger females and incidental lesions discovered during routine radiographic examination. The first patient, a 36-year-old female presented with an asymptomatic swelling and cortical expansion of the mandible adjacent to tooth #31. The second patient was a 13year-old female with an asymptomatic expansion in the left mandibular ramus. Lastly, a 15-year-old female with dull pain exhibited mild buccal and lingual bony expansion of the right mandible apical to tooth #18. Radiographically, two of the cases presented as multilocular radiolucent lesions and one demonstrated as a well-circumscribed unilocular radiolucency intermixed with radiopacities. All three lesions were treated with surgical curettage and no recurrence at one-year follow up. All cases exhibited sheets of foamy macrophages similar to other lytic xanthomatous lesions such as non-ossifying fibroma, benign fibrous histiocytoma, Rosai-Dorfman disease, Erdheim disease, Gaucher disease, etc. A diagnosis of CXJ was established by exclusion and involved careful correlation of the clinical, radiographic, histopathological and IHC features. All three cases revealed diffuse positivity for CD68 and were negative for S100. <h3>Conclusion</h3> CXJ are rare gnathic lesions of unknown etiology. We highlight three rare cases of CXJ, a lesion that may mimic other benign or reactive jaw lesions especially in young patients.