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

A 49-year-old man was admitted for medical examination because of epigastric discomfort. Esophagogastroscopy revealed a small, elevated, verrucoid pink-yellowish mucosal lesion, 3 mm in diameter, in the upper third of the esophagus ([Fig. 1]).

Fig. 1 Esophagogastroscopy showing a small, elevated, granular/verrucoid pink-yellowish lesion, 3 mm in diameter, in the mucosa of the upper third of the esophagus.

Low power histological examination revealed an exophytic lesion resembling a squamous papilloma, with the typical papillomatosis, acanthosis, and hyperparakeratosis of the esophageal squamous epithelium ([Fig. 2 a]).

Fig. 2 a Low power histological section showing papillomatosis, acanthosis, and hyperparakeratosis of the squamous epithelium, as usually seen in squamous papilloma (hematoxylin and eosin, original magnification × 40). b, c At higher magnification, the subepithelial connective tissue appears infiltrated by clear cells with foamy cytoplasm and small nuclei with no atypia (hematoxylin and eosin, original magnification: b × 200; c × 400). d There is diffuse and strong positivity for CD68 on immunostaining (original magnification × 400).

At higher magnification, neutrophilic intraepithelial exocytosis was observed, and the subepithelial connective tissue appeared infiltrated by clear cells with foamy cytoplasm and small nuclei with no atypia ([Fig. 2 b, c]). At immunohistochemistry, the foamy cells were negative for cytokeratins, s-100 protein, and CD1a, while CD68 was strongly positive ([Fig. 2 d]), 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]. The main 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].

Competing interests: None

Endoscopy_UCTN_Code_CCL_1AB_2AC_3AB

References

• 1 Shafer W. Verruciform xanthoma.  Oral Surg. 1971;  31 784-789
• 2 Iamaroon A, Vickers R A. Characterization of verruciform xanthoma by in situ hybridization and immunohistochemistry.  J Oral Pathol Med. 1996;  25 395-400
• 3 Khaskhely N M, Uezato H, Kamiyama T. et al . Association of human papillomavirus type 6 with a verruciform xanthoma.  Am J Dermatopathol. 2000;  22 447-452
• 4 Herrera-Goepfert R, Lizano-Soberón M, García-Perales M. Verruciform xanthoma of the esophagus.  Hum Pathol. 2003;  34 814-815