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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Verruciform xanthoma is a rare benign tumor first described by Shafer<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> in 1971&#46; It usually arises on the oral mucosa&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">An 11-year-old boy was referred to our outpatient clinic for evaluation of a lesion that had been present on the tongue for 6 weeks and was slightly tender on palpation&#46; The child had been diagnosed with acute myeloblastic leukemia 4 years earlier and had received 2 allogeneic bone marrow transplants&#44; the first 3 months after the diagnosis and the second 3 years later&#46; At the time we saw the patient&#44; he was in complete remission&#44; with full chimera of the transplant&#44; and he had never presented signs of graft-versus-host disease &#40;GVHD&#41;&#46; He had not received immunosuppressant treatment for the previous 7 months&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">Physical examination revealed an elevated&#44; oval-shaped&#44; sessile tumor with a long axis of about 2<span class="elsevierStyleHsp" style=""></span>cm&#46; The tumor was located on the left lateral surface of the tongue and had a whitish&#44; verrucous surface that was not ulcerated &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Two months before the lesion appeared&#44; asymptomatic reactivation of Epstein-Barr virus &#40;EBV&#41; infection had been detected&#44; evidenced by an increase in the viral load on serial measurements performed as part of the routine follow-up of the patient&#39;s hematologic disease&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Biopsy showed verrucous acanthosis with hyperkeratosis and isolated plaques of parakeratosis&#44; and there were numerous foamy histiocytes in the dermal papillae &#40;<a class="elsevierStyleCrossRefs" href="#fig0010">Figs&#46; 2 and 3</a>&#41;&#46; The lesion was diagnosed as verrucous xanthoma&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">The tumor was completely excised and there were no signs of recurrence during 2 months of follow-up&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Verrucous xanthoma usually presents clinically as a slow-growing asymptomatic lesion with a yellowish&#44; reddish&#44; or gray color&#44; a rough surface with a granular or papillomatous appearance&#44; a sessile or pedunculated base&#44; and it can measure up to 2<span class="elsevierStyleHsp" style=""></span>cm in diameter&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;3</span></a> The most common site is the oral mucosa&#44;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> although it can also arise on the genitalia<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;5</span></a> or&#44; more rarely&#44; on other sites of the skin&#46; Approximately 70&#37; of such tumors arising on the oral mucosa are located on the masticatory mucosa&#44; followed by the hard palate and the tongue&#44; as in our case&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> There have also been isolated cases of multifocal involvement of the skin and mucosas&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">With regard to the epidemiology of the tumor&#44; it can occur at any age&#44; though there is a slightly higher prevalence in middle age &#40;40-60 years&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Under 50 years of age&#44; the incidence is slightly higher in men whereas&#44; in the population over 50 years&#44; these tumors are slightly more common in women&#59; overall&#44; the frequency is practically the same in men and women&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The clinical differential diagnosis should include condyloma acuminatum&#44; common warts&#44; and squamous cell carcinoma &#40;including the verrucous form&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> In our case&#44; in view of the recent reactivation of EVB infection&#44; we also considered the possibility of lymphoma&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Histology of verrucous xanthoma reveals a hyperkeratotic squamous epithelium with focal parakeratosis and no cellular atypia&#59; foam cells are observed between elongated epithelial crests&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;6</span></a> These cells present a granular cytoplasm and the nucleus is typically eccentric&#46; Immunohistochemistry of these cells is intensely positive for CD68 and negative for S-100&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;3</span></a> Three histopathological patterns have been described&#58; verrucous&#44; papillary&#44; and flat&#46; The verrucous pattern is the most common&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">The pathological differential diagnosis includes the common wart&#44; lichen simplex chronicus&#44; and eruptive xanthoma&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> There have been no previous reports of an association between this tumor and infection with EBV&#46; However&#44; there have been descriptions of patients in whom this lesion has arisen on previous mucosal or cutaneous lesions&#44; such as oral lichen planus or mucosal GVHD&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Our patient did not have GVHD affecting the mucosa or any other previous lesions of the oral mucosa&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">The etiology and pathogenesis of verrucous xanthoma is not fully understood&#46; It has been suggested that it may be an inflammatory reaction secondary to recurrent trauma&#44; as the lesions mainly arise on areas subject to friction&#44;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> or that the accumulation of foam cells in the dermis is the primary trigger&#44; though no association with disorders of lipid metabolism has been found in the majority of the cases&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> No pathogenic role of the papillomavirus has been demonstrated&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> Given the known association between EBV and the induction of other lesions of the oral mucosa&#44; such as oral hairy leukoplakia&#44; in immunosuppressed patients&#44; we considered the possibility that the appearance of this lesion could have been related to reactivation of this virus in our patient&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> However&#44; no pathogenic relationship between EBV and verrucous xanthoma has been described to date&#44; and this could therefore be an incidental finding in our case&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">The treatment of choice is surgical excision of the lesion and recurrence is extremely rare&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">In summary&#44; we have presented the case of a patient with a lingual tumor having the typical clinical and histopathological features of verrucous xanthoma&#46; The patient presented recent reactivation of EBV infection&#44; an association not previously described in the literature and which could therefore be a casual finding&#46;</p></span>"
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Case and Research Letters
Verruciform Xanthoma Associated with Reactivation of Epstein-Barr Virus
Xantoma verruciforme asociado a reactivación de virus de Epstein-Barr
P. Maldonado-Cida,
Corresponding author
pmaldonadocid@gmail.com

Corresponding author.
, L. Noguera-Morela, M.J. Beato-Merinob, R. de Lucas-Lagunaa
a Servicio de Dermatología, Hospital Universitario La Paz, Madrid, Spain
b Servicio de Anatomía Patológica, Hospital Universitario La Paz, Madrid, Spain
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        "titulo" => "Xantoma verruciforme asociado a reactivaci&#243;n de virus de Epstein-Barr"
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Verruciform xanthoma is a rare benign tumor first described by Shafer<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> in 1971&#46; It usually arises on the oral mucosa&#46;</p><p id="par0010" class="elsevierStylePara elsevierViewall">An 11-year-old boy was referred to our outpatient clinic for evaluation of a lesion that had been present on the tongue for 6 weeks and was slightly tender on palpation&#46; The child had been diagnosed with acute myeloblastic leukemia 4 years earlier and had received 2 allogeneic bone marrow transplants&#44; the first 3 months after the diagnosis and the second 3 years later&#46; At the time we saw the patient&#44; he was in complete remission&#44; with full chimera of the transplant&#44; and he had never presented signs of graft-versus-host disease &#40;GVHD&#41;&#46; He had not received immunosuppressant treatment for the previous 7 months&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">Physical examination revealed an elevated&#44; oval-shaped&#44; sessile tumor with a long axis of about 2<span class="elsevierStyleHsp" style=""></span>cm&#46; The tumor was located on the left lateral surface of the tongue and had a whitish&#44; verrucous surface that was not ulcerated &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Two months before the lesion appeared&#44; asymptomatic reactivation of Epstein-Barr virus &#40;EBV&#41; infection had been detected&#44; evidenced by an increase in the viral load on serial measurements performed as part of the routine follow-up of the patient&#39;s hematologic disease&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">Biopsy showed verrucous acanthosis with hyperkeratosis and isolated plaques of parakeratosis&#44; and there were numerous foamy histiocytes in the dermal papillae &#40;<a class="elsevierStyleCrossRefs" href="#fig0010">Figs&#46; 2 and 3</a>&#41;&#46; The lesion was diagnosed as verrucous xanthoma&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0030" class="elsevierStylePara elsevierViewall">The tumor was completely excised and there were no signs of recurrence during 2 months of follow-up&#46;</p><p id="par0035" class="elsevierStylePara elsevierViewall">Verrucous xanthoma usually presents clinically as a slow-growing asymptomatic lesion with a yellowish&#44; reddish&#44; or gray color&#44; a rough surface with a granular or papillomatous appearance&#44; a sessile or pedunculated base&#44; and it can measure up to 2<span class="elsevierStyleHsp" style=""></span>cm in diameter&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;3</span></a> The most common site is the oral mucosa&#44;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> although it can also arise on the genitalia<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4&#44;5</span></a> or&#44; more rarely&#44; on other sites of the skin&#46; Approximately 70&#37; of such tumors arising on the oral mucosa are located on the masticatory mucosa&#44; followed by the hard palate and the tongue&#44; as in our case&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> There have also been isolated cases of multifocal involvement of the skin and mucosas&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">With regard to the epidemiology of the tumor&#44; it can occur at any age&#44; though there is a slightly higher prevalence in middle age &#40;40-60 years&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Under 50 years of age&#44; the incidence is slightly higher in men whereas&#44; in the population over 50 years&#44; these tumors are slightly more common in women&#59; overall&#44; the frequency is practically the same in men and women&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The clinical differential diagnosis should include condyloma acuminatum&#44; common warts&#44; and squamous cell carcinoma &#40;including the verrucous form&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> In our case&#44; in view of the recent reactivation of EVB infection&#44; we also considered the possibility of lymphoma&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Histology of verrucous xanthoma reveals a hyperkeratotic squamous epithelium with focal parakeratosis and no cellular atypia&#59; foam cells are observed between elongated epithelial crests&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;6</span></a> These cells present a granular cytoplasm and the nucleus is typically eccentric&#46; Immunohistochemistry of these cells is intensely positive for CD68 and negative for S-100&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;3</span></a> Three histopathological patterns have been described&#58; verrucous&#44; papillary&#44; and flat&#46; The verrucous pattern is the most common&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">The pathological differential diagnosis includes the common wart&#44; lichen simplex chronicus&#44; and eruptive xanthoma&#46;<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> There have been no previous reports of an association between this tumor and infection with EBV&#46; However&#44; there have been descriptions of patients in whom this lesion has arisen on previous mucosal or cutaneous lesions&#44; such as oral lichen planus or mucosal GVHD&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> Our patient did not have GVHD affecting the mucosa or any other previous lesions of the oral mucosa&#46;</p><p id="par0060" class="elsevierStylePara elsevierViewall">The etiology and pathogenesis of verrucous xanthoma is not fully understood&#46; It has been suggested that it may be an inflammatory reaction secondary to recurrent trauma&#44; as the lesions mainly arise on areas subject to friction&#44;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> or that the accumulation of foam cells in the dermis is the primary trigger&#44; though no association with disorders of lipid metabolism has been found in the majority of the cases&#46;<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">6</span></a> No pathogenic role of the papillomavirus has been demonstrated&#46;<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">9</span></a> Given the known association between EBV and the induction of other lesions of the oral mucosa&#44; such as oral hairy leukoplakia&#44; in immunosuppressed patients&#44; we considered the possibility that the appearance of this lesion could have been related to reactivation of this virus in our patient&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> However&#44; no pathogenic relationship between EBV and verrucous xanthoma has been described to date&#44; and this could therefore be an incidental finding in our case&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">The treatment of choice is surgical excision of the lesion and recurrence is extremely rare&#46;<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">In summary&#44; we have presented the case of a patient with a lingual tumor having the typical clinical and histopathological features of verrucous xanthoma&#46; The patient presented recent reactivation of EBV infection&#44; an association not previously described in the literature and which could therefore be a casual finding&#46;</p></span>"
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        "nota" => "<p class="elsevierStyleNotepara">Please cite this article as&#58; Maldonado-Cid P&#44; et al&#46; Xantoma verruciforme asociado a reactivaci&#243;n de virus de Epstein-Barr&#46; Actas Dermosifiliogr 2013&#59;104&#58;445&#8211;6&#46;</p>"
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Article information
ISSN: 15782190
Original language: English
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