Oral verruciform xanthoma and erythroplakia associated with chronic graft-versus-host disease: a rare case report and review of the literature

Abstract

BackgroundOral verruciform xanthoma is an uncommon benign lesion. Although oral verruciform xanthoma occurs in healthy individuals, it has been also reported in association with some inflammatory conditions. The aim of this study is to report a case of oral verruciform xanthoma associated with chronic graft-versus-host disease and to review the literature on this topic.Case presentationA 47-year-old Caucasian male presented to the Sector of Oral Medicine “V. Margiotta”, University Policlinic “P. Giaccone” of Palermo complaining of a mass on the gingiva. He first noticed the painless mass 1 year ago. He reported to have undergone allogenic hematopoietic stem cell transplantation 15 years ago for acute lymphoblastic leukaemia. Intraoral examination revealed a well-circumscribed, sessile yellowish and verrucous nodule upon canine, multiple yellowish and verrucous nodules on the hard palate, yellowish and verrucous nodules on left buccal mucosa. In addiction an area of white striae in a reticular pattern with erythema and ulceration was present on the dorsum of the tongue. This lesion was consistent with a known history of oral chronic graft versus host disease. Moreover, we observed a suspected area of oral erythroplakia yet on the dorsum of the tongue. In biopsy specimen of hard palate histopathological examination revealed a diagnosis of verrucous xanthoma of the oral cavity; in addiction in biopsy specimen of the dorsum of the tongue revealed the presence of erythroplakia with high grade dysplasia.ConclusionVerruciform xanthoma of the oral cavity associated with chronic graft-versus-host disease is a rare condition with a usually benign clinical course but malignant transformation has been described in association with oral potential malignant disorder (e.g. chronic graft versus host disease, erythroplakia). Very rare cases showed association with oral chronic graft versus-host-disease. To date, only eight cases were published in the world literature. Therefore it could be important follow up patients also for oral verruciform xanthoma onset.

Oral Lichen Planus: risk factors of malignant transformation and follow up. Ten years retrospective study

“…However, malignant transformation rate is a debated datum in the literature, especially because among studies there is no concordance in the definition of OLP diagnosis, specified by modified WHO diagnostic criteria (2003) ( 4 ). Furthermore, different PMDs can show similar clinical and histological features (OLL (24), Graft Versus Host Disease ( 25 )) accounTable for inaccurate diagnoses and consequently for bias in clinical studies.…”

A Snapshot of Knowledge about Oral Cancer in Italy: A 505 Person Survey

“…Interestingly, in our study, one third of participants believed that symptoms as burning or dry mouth were most indicative of oral cancer. Very few participants (13.86%) believed that red lesions might indicate OSCC and only 4.55% identified both white and red plaque; this is particularly impressive, given the high rate of malignant transformation reported in red oral plaque (erythroplakias) [ 35 , 36 ], As a consequence of this misunderstanding of initial signs, patients may not consult a dentist, increasing the duration of diagnostic delay.…”

Verruciform xanthoma associated with lichen planus

“…16 In PubMed, only 16 cases have been reported, associating VX with OLP in the oral mucosa, including the present case. 6,8,10,12,15,[17][18][19][20][21][22] It stands out that the studies of Yu et al, 10 Ide et al 15 and Andrade et al, 6 did not present data about the demographic, clinical, or histologic characteristics of the VX and OLP. For this reason, these three articles were not included in Table 1.…”

“…3,5,11 Its pathogenesis is still unclear, although reports of occurrence associated with lichen planus and other immunological and autoimmune diseases are noted, such as pemphigus vulgaris, lupus erythematosus, dystrophic bullous epidermolysis and graft versus host disease, especially when on the skin. 3,8,10,12 Rare cases of VX associated with oral lichen planus (OLP) in the oral mucosa have been confirmed through clinical and histopathological examinations. Therefore, present study aims to report a clinical case of VX associated with OLP, addressing clinical, histopathological and immunohistochemical aspects to obtain data to aid in the understanding of its pathogenesis.…”