Unusual presentation of primary central xanthoma of the maxilla associated with impacted canine: An update on immunohistochemistry in the diagnosis

Abstract

A xanthoma is an extremely rare condition that affects the soft tissues and bones and is characterized by a predominance of lipid-rich foamy histiocytes. The onset of xanthomas is frequently accompanied by primary or secondary hyperlipidemia. Primary bone xanthomas are very uncommon benign bone lesions that are not linked to hyperlipidemia. Histopathologically, they are distinguished by histiocytes, an abundance of foam cells or xanthoma cells that contain lipids, and a paucity of multinucleated giant cells. There have only been four reports of primary maxillary xanthoma in the medical literature. We present a rare primary intrabony xanthoma of the anterior maxilla in a 23-year-old normolipidemic female patient with solitary radiolucency. Using CD68, S-100, and CD1a immunohistochemical staining, it is possible to distinguish between macrophage/non-Langerhans histiocytes and Langerhans histiocytes. Therefore, a diagnosis of a central xanthoma of the jaws must be made.

Keywords: Bone; CD68; foamy histiocytes; maxilla; non-Langerhans histiocyte; xanthoma.

Figure 1

CT shows a well-defined, circumscribed, and low-density lesion at the right anterior maxilla with impacted teeth and mild displacement of the first premolar and lateral incisor

Figure 2

Low power magnification (100×) HE stained tissue section showing numerous xanthoma cells in a background of loose fibrous stroma

Figure 3

High magnification (450×) HE stained tissue section demonstrating the abundant xanthoma cells showing distinct cytoplasmic membranes with granular cytoplasm and small, hyperchromatic nuclei

Figure 4

Xanthoma cells exhibiting strong positive CD68 IHC staining and negative for S-100 and CD1a IHC staining