Verruciform xanthoma is a benign mucocutaneous, uncommon, nonsymptomatic lesion of uncertain etiopathology, which occurs mostly on the oral mucosa of middle-aged individuals. Histopathologically, VX is diagnosed by presence of lipid-laden foam cells in papillary region of connective tissue. A 60-year-old male patient presented with a painless growth on the left buccal mucosa. On clinical examination a yellowish white exophytic lesion, measuring 11 × 7？mm in size, was found, which was cauliflower-shaped on inspection and painless on palpation. Histopathological examination revealed varying degrees of surface parakeratosis and the accumulation of numerous foam cells in the connective tissue papillae among the uniformly elongated epithelial ridges. On immunohistochemical staining, there was a neutrophilic infiltrate of the epidermis with CD68 positive xanthoma cells restricted to the papillary dermis, mixed with other chronic inflammatory cells. 1. Introduction Verruciform xanthoma (VX) is an uncommon benign mucocutaneous lesion that resembles virus-induced papilloma but has an unknown etiology and uncertain nature. It was first reported by Shafer in 1971. VX usually occurs in the oral mucosa of the middle-aged individuals. It most commonly presents with a verrucous appearance. However in some instances it may appear polypoid, papillomatous, or sessile. It occurs as a small (0.2–2？cm), solitary, asymptomatic, slow growing, white or yellowish red lesion with no sex predilection [1, 2]. Histologically VX is distinguished from other lesions by the presence of large numbers of foam cells in, and essentially limited to, the connective tissue papillae. The foam cells on ultrastructural studies have been concluded to be fat-laden macrophages [3, 4]. Other cell types, including Langerhans cells, intraepithelial neutrophilic infiltrate, and even fibroblasts, have been reported [1–3]. A variable degree of parakeratosis is observed which is present in the crypts between papillae which are of variable length and thickness, often extending close to the surface. The rete pegs are extremely elongated and uniform [2]. Almost every VX case is diagnosed on histological examination as the clinical appearance is not diagnostic. Differentiation from other lesions with foamy or granular cells is not difficult as the VX is the only lesion to have these cells confined to the papillae [4, 5]. Differential diagnosis includes erythroplasia of Queyrat (Bowen disease of the glans penis), seborrheic keratosis, verrucous carcinoma, verruca simplex, and condyloma acuminatum [6, 7]. The
References
[1]
S. K. Mohsin, M. W. Lee, M. B. Amin et al., “Cutaneous verruciform xanthoma: a report of five cases investigating the etiology and nature of xanthomatous cells,” The American Journal of Surgical Pathology, vol. 22, no. 4, pp. 479–487, 1998.
[2]
R. Rajendran, Shafer's Textbook of Oral Pathology, 6th edition, 2009.
[3]
A. Buchner, L. S. Hansen, and P. W. Merrell, “Verruciform xanthoma of the oral mucosa: report of five cases and review of the literature,” Archives of Dermatology, vol. 117, no. 9, pp. 563–565, 1981.
[4]
W. G. Shafer, “Verruciform xanthoma,” Oral Surgery, Oral Medicine, Oral Pathology, vol. 31, no. 6, pp. 784–789, 1971.
[5]
T. Xia, G. Li, I. S. Jun, Y. N. Yinglu Guo, and M. K. Chung, “Verruciform xanthoma of the penis,” Korean Journal of Urology, vol. 45, pp. 297–298, 2004.
[6]
O. Reich and S. Regauer, “Recurrent verruciform xanthoma of the vulva,” International Journal of Gynecological Pathology, vol. 23, no. 1, pp. 75–77, 2004.
[7]
S. S. Farahani, N. S. Treister, Z. Khan, and S.-B. Woo, “Oral verruciform xanthoma associated with chronic graft-versus-host disease: a report of five cases and a review of the literature,” Head and Neck Pathology, vol. 5, no. 2, pp. 193–198, 2011.
[8]
N. M. Khaskhely, H. Uezato, T. Kamiyama, et al., “Association of human papillomavirus type 6 with a verruciform xanthoma,” The American Journal of Dermatopathology, vol. 22, no. 5, pp. 447–452, 2000.
[9]
S. K. Mohsin, M. W. Lee, M. B. Amin, et al., “Cutaneous verruciform xanthomna: a report of five cases investigating the etiology and nature of xanthomatous cells,” The American Journal of Surgical Pathology, vol. 22, pp. 479–487, 1998.
[10]
S. Y. Rawal, J. R. Kalmar, and D. N. Tatakis, “Verruciform xanthoma: immunohistochemical characterization of xanthoma cell phenotypes,” Journal of Periodontology, vol. 78, no. 3, pp. 504–509, 2007.
[11]
H. P. Philipsen, P. A. Reichart, T. Takata, and I. Ogawa, “Verruciform xanthoma: biological profile of 282 oral lesions based on a literature survey with nine new cases from Japan,” Oral Oncology, vol. 39, no. 4, pp. 325–336, 2003.
[12]
S. S. Farahani, N. S. Treister, Z. Khan, and S.-B. Woo, “Oral verruciform xanthoma associated with chronic graft-versus-host disease: a report of five cases and a review of the literature,” Head and Neck Pathology, vol. 5, no. 2, pp. 193–198, 2011.
[13]
D. J. Zegarelli, E. C. Zegarelli Schmidt, and E. V. Zegarelli, “Verruciform xanthoma: further light and electron microscopic studies, with the addition of a third case,” Oral Surgery Oral Medicine and Oral Pathology, vol. 40, no. 2, pp. 246–256, 1975.
[14]
V. Raphael, H. Das, R. Sarma, and B. Shunyu, “Oral verruciform xanthoma: a case report,” International Journal of Oral and Maxillofacial Pathology, vol. 3, no. 2, pp. 65–67, 2012.
[15]
R. Herrera-Goepfert, M. Lizano-Soberón, and M. Garcia-Perales, “Verruciform xanthoma of the esophagus,” Human Pathology, vol. 34, no. 8, pp. 814–815, 2003.
[16]
C.-H. Yu, T.-C. Tsai, J.-T. Wang et al., “Oral verruciform xanthoma: a clinicopathologic study of 15 cases,” Journal of the Formosan Medical Association, vol. 106, no. 2, pp. 141–147, 2007.
[17]
H. P. Philipsen, P. A. Reichart, T. Takata, and I. Ogawa, “Verruciform xanthoma: biological profile of 282 oral lesions based on a literature survey with nine new cases from Japan,” Oral Oncology, vol. 39, no. 4, pp. 325–336, 2003.
[18]
E. W. Odell and P. R. Morgan, Eds., Biopsy Pathology of the Oral Tissue, Chapman Hall, London, UK, 1st edition, 1998.
[19]
S. Kimura, “Verruciform xanthoma of the scrotum,” Archives of Dermatology, vol. 120, no. 10, pp. 1378–1379, 1984.
[20]
K. A. Mostafa, T. Takata, I. Ogawa, N. Ijuhin, and H. Nikai, “Verruciform xanthoma of the oral mucosa: a clinicopathological study with immunohistochemical findings relating to pathogenesis,” Virchows Archiv, vol. 423, no. 4, pp. 243–248, 1993.
[21]
S. B. Connolly, E. J. Lewis, J. S. Lindholm, B. D. Zelickson, C. B. Zachary, and W. D. Tope, “Management of cutaneous verruciform xanthoma,” Journal of the American Academy of Dermatology, vol. 42, no. 2, pp. 343–347, 2000.
