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      Oral Verruciform Xanthoma: A Case Report and Literature Review

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          Abstract

          Oral verruciform xanthoma represents an uncommon entity, which affects mainly oral mucosa. This paper presents the major clinical and histological features of oral verruciform xanthoma and reports a case on the tongue. The differential diagnosis and a literature review are also provided in light of recent information.

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          Verruciform xanthoma--biological profile of 282 oral lesions based on a literature survey with nine new cases from Japan.

          H.P Philipsen, P.A Reichart, T Takata … (2003)
          The biological profile of oral verruciform xanthoma (VX) is presented based on a world-wide literature survey of 282 cases. From 1979 onwards, extraoral cases have also been reported. This rare, harmless lesion with a sessile or pedunculated base is a red/pink, papillary/granular/verrucous mucosal growth, occurring in females (mean age, 54.9 yrs) and males (mean age, 44.2 yrs) in a female:male ratio of 1:1.1. The most common location is by far the gingival margin and other areas of the masticatory oral mucosa. Comparison between 173 non-Japanese and 109 Japanese patients with oral VX showed few discrepancies in epidemiological data, indicating only few significant ethnic differences between the two cohorts. Histomorphologically, the epithelium covering the lesion can be divided into three groups: (A) a verrucous, (B) a papillary and (C) a flat pattern. The hallmark of all VX, irrespective of the lesion being intra- or extraoral is, however, the presence of vacuolated, foam or xanthoma cells which ultimately replace the connective tissue between the epithelial ridges. The xanthoma cells have been shown to be cells of the monocyte/macrophage lineage. The present concept of the etiology and pathogenesis of VX, including the possible viral (HPV) association is revised, based on both intra- and some extraoral cases, and it is concluded that it is still far from being clarified. Copyright 2002 Elsevier Science Ltd.
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            Verruciform xanthoma of the oral mucosa. Report of four cases and a review of the literature.

            Beate R. Jaeger, Adrian Cabral, Alex M. Jaeger … (2001)
            We present four new cases of verruciform xanthoma (VX) in the oral mucosa and review the literature. Clinical, histological, and immunohistochemical features of four new cases of VX were analysed together with cases found in a review of the literature. Expression of CD-68 was studied by immunohistochemistry. Only 162 cases were reported in the oral mucosa. Ninety were males (55.5%) and 72 were females (44.5%). Mean age was 44.9 years. The majority of cases occurred in masticatory mucosa (69.7%). Our cases exhibited papillary or verrucous proliferation of squamous epithelium associated with hyperparakeratosis and with numerous foamy cells confined to the lamina propria papillae. Foamy cells were positive to CD-68 antibody, showing a macrophagic nature. VX is a rare benign lesion, and is probably inflammatory. However, its aetiology and pathological mechanisms remain unknown.
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              Cellular basis of verruciform xanthoma: immunohistochemical and ultrastructural characterization.

              K Kusama, Hiromu Mishima, Anneyuko Saito … (2008)
              Verruciform xanthoma (VX) holds two basic pathogenic interests: (1) Why and how do macrophage foam cells accumulate exclusively in the sub-basal papillae? and (2) What underlies the disease chronicity? Moreover, an unsolved question is which came first - epithelial hyperplasia or foam cell collection? We analyzed 36 oral mucosal lesions to dissect a series of linked cellular changes in VX using immunohistochemical and ultrastructural techniques. Macrophage scavenger receptor-1 (MSR-1), monocyte chemoattractant protein-1 (MCP-1), CCR2, and oxidized low-density lipoprotein (ox-LDL) were all expressed by foam cells. VX epithelium showed reactivity for MCP-1, HLA-DR and IL8 in varying degrees, and showed a nearly 40% reduction in Langerhans cell density. In sub-epithelial inflammatory infiltrates, CD8+ T cells preponderated (>70%), but only a minority were positive for granzyme B (<1%). Keratinocyte/basal lamina complex exhibited disruption of basal lamina, squamatization and cytolysis of basal cells, fragmentation of desmosomes, and intraepithelial migration of macrophages. In severely inflamed papillae, necrotic foam cells were scavenged by adjacent macrophages. Under synergistic regulation of T cells, MCP-1/CCR2-mediated macrophage recruitment in the sub-basal papillae and the lysosomal engulfment of epithelial lipids by MSR-1-bearing macrophages may be central in VX formation. Once developed, ox-LDL-induced foam cell necrosis and macrophage-dependent debris disposal may cyclically perpetuate VX.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Case Rep Pathol
                Journal ID (iso-abbrev): Case Rep Pathol
                Journal ID (publisher-id): CRIPA
                Title: Case Reports in Pathology
                Publisher: Hindawi Publishing Corporation
                ISSN (Print): 2090-6781
                ISSN (Electronic): 2090-679X
                Publication date (Print): 2014
                Publication date (Electronic): 8 December 2014
                Volume: 2014
                Electronic Location Identifier: 641015
                Affiliations
                1Department of Bioscience and Oral Diagnosis, Dental School, Institute of Science and Technology (ICT), Paulista State University (UNESP), 777 Engenheiro Francisco José Longo Avenue, 12245-000 São José dos Campos, SP, Brazil
                2Department of Physiology and Pathology, Paulista State University (UNESP), 1680 Humaitá Street, 14801-903 Araraquara, SP, Brazil
                3Department of Oral Pathology, Universitty of São Paulo (USP), 2227 Professor Lineu Prestes Avenue, 05508-000 São Paulo, SP, Brazil
                4Department of Diagnosis and Surgery, Paulista State University (UNESP), 1680 Humaitá Street, 14801-903 Araraquara, SP, Brazil
                Author notes
                *Yonara Maria Freire Soares Marques: yonarafreire@ 123456yahoo.com.br

                Academic Editor: Yoji Nagashima

                Author information
                http://orcid.org/0000-0001-7015-7175
                Article
                DOI: 10.1155/2014/641015
                PMC ID: 4274645
                PubMed ID: 25548705
                SO-VID: ebca930d-a307-4adf-883e-67ccdbfc65c4
                Copyright © Copyright © 2014 Yonara Maria Freire Soares Marques et al.
                License:

                This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                History
                Date received : 22 July 2014
                Date accepted : 24 November 2014
                Categories
                Subject: Case Report

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