Autopsy and Case Reports
https://www.autopsyandcasereports.org/article/doi/10.4322/acr.2021.360
Autopsy and Case Reports
Clinical Case Report and Review

Verruciform xanthoma associated with lichen planus

Cristianne Kalinne Santos Medeiros; Glória Maria de França; Weslay Rodrigues da Silva; Joaquim Felipe Júnior; Hébel Cavalcanti Galvão; Patrícia Teixeira de Oliveira

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Abstract

Verruciform xanthoma (VX) is a rare benign lesion of unknown etiology, with a rough or papillary aspect, painless, sessile, well-defined, most lesions do not exceed 2 cm in their largest diameter, the degree of keratinization of the surface influences color, varying white to red, affecting mainly the gingiva and alveolar mucosa, and can also be seen in skin and genital. Herein, we present a report a clinical case of oral verruciform xanthoma in the buccal mucosa associated with the lichen planus lesion, as well as the morphological and immunohistochemical characteristics of the lesion. The clinical diagnostic hypothesis of oral lichen planus of the white reticular lesions on the buccal mucosa and on the tongue was confirmed by histopathology before a subepithelial connective tissue exhibiting intense inflammatory infiltrate in a predominantly lymphocytic band. In contrast, the hypothesis of the verrucous lesion in the left buccal mucosa was leukoplakia, with histopathological evidence showing exophytic and digitiform proliferations with parakeratin plugs between the papillary projections. Subepithelial connective tissue was characterized by macrophages with foamy cytoplasm (xanthoma cells). An immunohistochemical examination was performed, showing positivity for CD68, a macrophage marker, in addition to testing by Schiff's periodic acid (PAS) with diastasis, which was detected the presence of lipids inside these macrophages. The patient is free of recurrences of verruciform xanthoma and is being monitored due to the presence of lesions of oral lichen planus.

Keywords

Foam cells, Mouth Mucosa, Pathology

References

1 Shafer WG. Verruciform xanthoma. Oral Surg Oral Med Oral Pathol. 1971;31(6):784-9. http://dx.doi.org/10.1016/0030-4220(71)90134-4. PMid:5280461.

2 Farahani SS, Treister NS, Khan Z, Woo SB. Oral verruciform xanthoma associated with chronic graft-versus- host disease: a report of five cases and a review of the literature. Head Neck Pathol. 2011;5(2):193-8. http://dx.doi.org/10.1007/s12105-011-0246-2. PMid:21305367.

3 Tamiolakis P, Theofilou VL, Tosios KI, Sklavounou-Andrikopoulou A. Oral verruciform xanthoma: report of 13 new cases and review of the literature. Med Oral Patol Oral Cir Bucal. 2018;23(4):e429-35. http://dx.doi.org/10.4317/medoral.22342. PMid:29924759.

4 Gannepalli A, Appala A, Reddy L, Babu DBG. Insight into verruciform xanthoma with oral submucous fibrosis: case report and review of literature. J Oral Maxillofac Pathol. 2019;23(4, Suppl 1):43-8. http://dx.doi.org/10.4103/jomfp.JOMFP_210_18. PMid:30967723.

5 Philipsen HP, Reichart PA, Takata T, Ogawa I. Verruciform xanthoma-biological profile of 282 oral lesions based on a literature survey with nine new cases from Japan. Oral Oncol. 2003;39(4):325-36. http://dx.doi.org/10.1016/S1368-8375(02)00088-X. PMid:12676251.

6 Andrade BAB, Agostini M, Pires FR, et al. Oral verruciform xanthoma: a clinicopathologic and immunohistochemical study of 20 cases. J Cutan Pathol. 2015;42(7):489-95. http://dx.doi.org/10.1111/cup.12500. PMid:25930961.

7 Cebeci F, Verim A, Somay A, Çalıkoğlu E. Verruciform xanthoma of a lower lip lesion: a new case and review of the literature. Case Rep Dermatol. 2017;9(2):130-5. http://dx.doi.org/10.1159/000477961. PMid:29033816.

8 Theofilou VI, Sklavounou A, Argyris PP, Chrysomali E. Oral verruciform xanthoma within lichen planus: a case report and literature review. Case Rep Dent. 2018;2018:1615086. http://dx.doi.org/10.1155/2018/1615086. PMid:29850274.

9 Belknap AN, Islam MN, Bhattacharyya I, Cohen DM, Fitzpatrick SG. Oral verruciform xanthoma: a series of 212 cases and review of the literature. Head Neck Pathol. 2020;14(3):742-8. http://dx.doi.org/10.1007/s12105-019-01123-0. PMid:31898056.

10 Yu CH, Tsai TC, Wang JT, et al. Oral verruciform xanthoma: a clinicopathologic study of 15 cases. J Formos Med Assoc. 2007;106(2):141-7. http://dx.doi.org/10.1016/S0929-6646(09)60230-8. PMid:17339158.

11 Mainville GN. Non-HPV papillary lesions of the oral mucosa: clinical and histopathologic features of reactive and neoplastic conditions. Head Neck Pathol. 2019;13(1):71-9. http://dx.doi.org/10.1007/s12105-019-01001-9. PMid:30693457.

12 Capocasale G, Panzarella V, Tozzo P, et al. Oral verruciform xanthoma and erythroplakia associated with chronic graft-versus-host disease: a rare case report and review of the literature. BMC Res Notes. 2017;10(1):631. http://dx.doi.org/10.1186/s13104-017-2952-7. PMid:29183344.

13 Zegarelli DJ, Aegarelli-Schmidt EC, Zegarelli EV. Verruciform xanthoma. A clinical, light microscopic, and electron microscopic study of two cases. Oral Surg Oral Med Oral Pathol. 1974;38(5):725-34. http://dx.doi.org/10.1016/0030-4220(74)90393-4. PMid:4373690.

14 Byakodi S, Kumar B, Patil S, Shinde S. Verruciform xanthoma of the tongue. Natl J Maxillofac Surg. 2017;8(1):78-80. http://dx.doi.org/10.4103/0975-5950.208982. PMid:28761282.

15 Ide F, Obara K, Yamada H, Mishima K, Saito I, Kusama K. Cellular basis of verruciform xanthoma: immunohistochemical and ultrastructural characterization. Oral Dis. 2008;14(2):150-7. http://dx.doi.org/10.1111/j.1601-0825.2007.01362.x. PMid:18302675.

16 Fite C, Plantier F, Dupin N, Avril MF, Moyal-Barracco M. Vulvar verruciform xanthoma: ten cases associated with lichen sclerosus, lichen planus, or other conditions. Arch Dermatol. 2011;147(9):1087-92. http://dx.doi.org/10.1001/archdermatol.2011.113. PMid:21576553.

17 Neville BW, Weathers DR. Verruciform xanthoma. Oral Surg Oral Med Oral Pathol. 1980;49(5):429-34. http://dx.doi.org/10.1016/0030-4220(80)90287-X. PMid:6154914.

18 Hume WJ, Smith CJ, Franklin CD. Verruciform xanthoma. Br J Oral Surg. 1980;18(2):157-61. http://dx.doi.org/10.1016/0007-117X(80)90034-7. PMid:6934808.

19 Miyamoto Y, Nagayama M, Hayashi Y. Verruciform xanthoma occurring within oral lichen planus. J Oral Pathol Med. 1996;25(4):188-91. http://dx.doi.org/10.1111/j.1600-0714.1996.tb00218.x. PMid:8809688.

20 Polonowita AD, Firth NA, Rich AM. Verruciform xanthoma and concomitant lichen planus of the oral mucosa. A report of three cases. Int J Oral Maxillofac Surg. 1999;28(1):62-6. http://dx.doi.org/10.1016/S0901-5027(99)80682-1. PMid:10065655.

21 Anbinder AL, Quirino MR, Brandao AA. Verruciform xanthoma and neurofibromatosis: a case report. Br J Oral Maxillofac Surg. 2011;49(4):e6-7. http://dx.doi.org/10.1016/j.bjoms.2010.06.013. PMid:20638163.

22 Barrett AW, Boyapati RP, Bisase BS, et al. Verruciform xanthoma of the oral mucosa: a series of eight typical and three anomalous cases. Int J Surg Pathol. 2019;27(5):492-8. http://dx.doi.org/10.1177/1066896919827374. PMid:30727785.

23 Nowparast B, Howell FV, Rick GM. Verruciform xanthoma: a clinicpathologic review and report of fifty-four cases. Oral Surg Oral Med Oral Pathol. 1981;51(6):619-25. http://dx.doi.org/10.1016/S0030-4220(81)80012-6. PMid:6942361.
 


Submitted date:
02/02/2021

Reviewed date:
04/13/2021

Publication date:
02/21/2022

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