Primary Xanthoma of the Mandible: Report of a Rare Case

Abstract

Xanthoma is a lesion most commonly seen in soft tissues such as the skin, subcutis, or tendon sheaths. Xanthoma formation is often associated with primary or secondary hyperlipidemia. Primary bone xanthomas are extremely rare benign bone lesions not associated with hyperlipidemia, histopathologically characterized by histiocytes, abundant lipid containing macrophages (foam cells), and multinucleated giant cells. Cholesterol clefts can be found in the medullary bone. Less than ten cases of xanthoma in the mandible have been reported. We present a rare primary intrabony xanthoma in a normolipidemic patient.

Intraosseous mandibular xanthomas – an interesting diagnostic dilemma?

“…No definite reason can be found to justify this recurrence; however, it is possible that inadequate initial treatment or persistence of the unidentified aetiological process may have led to further extravasation of foamy macrophages, precipitating recurrence of the xanthoma. In addition, the finding of the non‐vital lower right first molar may also be implicated due to the theoretical reparative–regenerative aetiology of intraosseous xanthomas, which has been discussed by previous authors. Although the majority of reported cases do not discuss dental history, several cases within the literature report potential sources of a reactive–reparative aetiology, including endodontic work (2 cases), previous surgery (2 cases) or orthodontics (3 cases) in the region of the xanthoma (Table ).…”

“…In all other cases within the literature, surgical excision of the gnathic xanthoma resulted in resolution of the lesion. In a recent paper, partial excision has been advocated as satisfactory treatment, allowing for regression of the lesion; however, due to the possibility of recurrence highlighted by this case, more definitive treatment is recommended. This is further highlighted by the lack of resolution (and in some cases, increase in lesion size), found in the xanthoma case series by Harsanyi and Larsson, where patients were treated with an incisional biopsy only.…”

“…Compression of neurological structures may result in symptoms – as found in cases of intracranial xanthomas – but the lesion is otherwise considered asymptomatic. There are three reported cases of a maxillary intraosseous xanthoma within the literature and several cases of mandibular involvement, none of which report recurrence (see Table ). No malignant transformation of intraosseous xanthomas has been reported.…”

Intraosseous xanthoma of the mandible: A multi‐institutional case series with a literature review

“…Thus far, few cases of IOX of the jaws with enough clinicopathologic and radiographic details to confirm the diagnosis have been documented in the literature . Since case series of IOX of the jaws are infrequent in the literature, in order to provide consistent and well‐documented reports that may serve as a basis for a better understanding of the lesion, we describe here a series of five new cases of IOX of the mandible in a multi‐institutional study, with emphasis on clinicopathological, radiographic and immunohistochemical data of this condition as compared with reports detected in a literature review.…”

“…This condition typically occurs in endocrine or metabolic diseases with increased cholesterol levels, in particular primary hyperlipidaemia, hypolipoproteinaemia type II and III, diabetes mellitus, hypothyroidism and lipoprotein lipase deficiency, and therefore, it is called secondary bone xanthoma . In the absence of systemic diseases, the lesion is called primary xanthoma …”