Verruciform xanthoma of the esophagus: an uncommon entity in an unusual site.

ical examination because of epigastric discomfort. Esophagogastroscopy revealed a small, elevated, verrucoid pink-yellowish mucosal lesion, 3mm in diameter, in the upper third of the esophagus (●" Fig. 1). Low power histological examination revealed an exophytic lesion resembling a squamouspapilloma,with the typical papillomatosis, acanthosis, and hyperparakeratosis of the esophageal squamous epithelium (●" Fig. 2a). At higher magnification, neutrophilic intraepithelial exocytosis was observed, and the subepithelial connective tissue appeared infiltrated byclear cellswith foamy cytoplasm and small nuclei with no atypia (●" Fig. 2b,c). At immunohistochemistry, the foamycellswere negative for cytokeratins, s-100 protein, and CD1a, while CD68 was strongly positive (●" Fig. 2d), indicating the histiocytic nature of the cells. The histological and immunohistochemical features allowed making a diagnosis of verruciform xanthoma of the esophagus. Verruciform xanthoma is a lesion characteristically described in the oral cavity and genital skin [1]. It is usually solitary, but cases of multifocal lesions have been reported [1]. Themain histological feature is the presence of foamy histiocytes in the subepithelial stroma of a squamous epithelium displaying papillomatosis, acanthosis, and hyperkeratosis, as observed in papillomatous/verrucous lesions. Intraepithelial neutrophilic infiltration is another hallmark. The etiology is still unknown, most cases being unrelated to a viral infection. The presence of human papilloma virus in the epithelial cells has been demonstrated in only two reported lesions in the oral mucosa [2] and scrotum [3]. To the best of our knowledge, our case represents the second description in the English literature of verruciform xanthoma in the esophagus [4].