Verruciform xanthoma of the oral cavity is a rare benign lesion. Histologically, it is characterized by papillary or verrucous proliferation of squamous epithelium and numerous foam cells. This disease was first described by Shafer in 1971. We present a very rare case of a verruciform xanthoma with epithelial dysplasia arising in the buccal mucosa of a 68-year-old Japanese man. The patient referred to our clinic because of a mass in the left buccal mucosa. The lesion was white, and its surface was granulated. The patient had neither tenderness nor any symptoms. The lesion was clinically diagnosed to be a benign tumor, and biopsy was performed. Histopathologically, the tumor consisted of stratified squamous epithelium with dysplasia, hyperchromatism, and parakeratosis and showed elongated rete ridges. Between and under the rete ridges, aggregation of foam cells was observed. Immunohistochemically, cells stained positively for PCNA and Ki-67 in the squamous epithelium with dysplasia. The lesion was diagnosed to be a verruciform xanthoma with epithelial dysplasia. Under local anesthesia, the lesion was excised. The patient has remained free of local recurrence for 2 years after surgery.
