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the floor of the mouth&#44; and the lateral edges and ventral region of the tongue &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; The rest of the physical examination was normal&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Histopathology</span><p id="par0015" class="elsevierStylePara elsevierViewall">An incisional biopsy of the buccal mucosa was taken&#46; Staining with hematoxylin&#8211;eosin revealed a squamous mucosa with acanthosis and clear cells corresponding to keratinocytes with abundant clear cytoplasm and a small nucleus and foci of a perinuclear cytoplasmic eosinophilic material&#46; The cell wall showed no inflammatory infiltrate &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; PAS staining revealed no mycotic structures&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Other Tests</span><p id="par0020" class="elsevierStylePara elsevierViewall">Mycology culture was negative&#46; Blood tests revealed elevated urea &#40;356&#8239;mg&#47;dL&#41;&#44; and other biochemistry parameters&#44; blood count&#44; complement&#44; and immunoglobulin levels were normal&#46; The serologic study &#40;EBV&#44; <span class="elsevierStyleItalic">Mycoplasma pneumoniae</span>&#44; parvovirus B19&#44; CMV&#41; showed findings compatible with past infection &#40;IgG positive&#41; by EBV and Parvovirus B19&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">&#91;&#91;&#63;&#93;&#93;What is your diagnosis&#63;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Diagnosis</span><p id="par0030" class="elsevierStylePara elsevierViewall">White sponge nevus&#46;</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinical Course and Treatment</span><p id="par0035" class="elsevierStylePara elsevierViewall">A wait-and-see approach was adopted due to the benign nature of this entity&#46; The patient is currently being studied for high urea levels at a metabolic diseases department&#46;</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Comment</span><p id="par0040" class="elsevierStylePara elsevierViewall">White sponge nevus is a rare&#44; autosomal dominant hereditary genodermatosis with variable penetrance&#44; although sporadic cases have been reported&#46; It was described by Hyde in 1909 with the name leukokeratosis and in 1935&#44; Cannon coined the term <span class="elsevierStyleItalic">white sponge nevus</span>&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">The pathophysiology is not fully understood&#46; Mutations have been identified in the genes that code for keratins 3 and 14&#44; located on chromosomes 12q13 and 17q21&#8208;q22&#44; respectively&#46; These keratins are expressed specifically in the buccal&#44; anogenital&#44; nasal&#44; esophageal&#44; and laryngeal mucosa&#44; and in other nonkeratinized stratified squamous epithelia&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;4</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Lesions are asymptomatic in most cases and tend to involve the buccal mucosa bilaterally and symmetrically&#44; in the form of white plaques with a rough&#44; spongy appearance&#44; which do not detach on scraping&#46; Rare cases of isolated involvement of the extraoral mucosa have been reported&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> They may be congenital or appear during childhood&#44; as in the case of our patient&#46; They sometimes appear during adolescence and show no preference for sex&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3&#44;5&#44;6</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Histopathology is nonspecific&#46; Of note is the epithelial thickening with parakeratotic hyperkeratosis&#44; acanthosis&#44; and spongiosis&#44; with vacuolization of supra-epidermal keratinocytes and with perinuclear eosinophilic aggregates that correspond to anomalous groupings of keratin tonofilaments&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Diagnosis is based on clinical signs and symptoms and on histopathology findings&#46; A genetic study may also be performed&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">Prognosis is good&#46; Cases have been reported in the literature with subsequent development of dysplasia and even epidermoid carcinoma&#44; although the link has not been clearly established&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;5</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">Treatment is not required&#44; as the lesions are asymptomatic&#46; Isolated cases of response to chlorhexidine&#44; topical retinoids and topical tetracycline&#44; and oral penicillin have been published&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2&#44;6</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">The differential diagnosis must be carried out with other diseases that involve white lesions in the buccal mucosa&#44; such as genodermatosis &#40;congenital pachyonychia&#44; hereditary benign intraepithelial dyskeratosis&#44; and congenital dyskeratosis&#41;&#44; infections &#40;oral candidiasis in its pseudomembranous form&#44; focal epithelial hyperplasia&#44; and syphilis&#41;&#44; inflammatory diseases &#40;lichen planus&#41;&#44; premalignant and malignant lesions &#40;leukoplasia&#44; epidermoid carcinoma&#41;&#44; and other entities such as leukoedema and friction hyperkeratosis&#46;</p><p id="par0080" class="elsevierStylePara elsevierViewall">We report a rare&#44; extensive case of pediatric-onset white sponge nevus&#46; Of note in our patient is the casual finding of high levels of urea and suspected underlying metabolic disease&#44; a finding not reported to date&#46; The link between the 2 entities has yet to be defined&#46;</p></span><span id="sec0040" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0040">Conflicts of Interest</span><p id="par0085" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest&#46;</p></span></span>"
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Case for Diagnosis
White Plaques in the Oral Cavity
Placas blancas en cavidad oral
E. Rojas Moraa,
Corresponding author
ester.rojas@salud.madrid.org

Corresponding author.
, R. Kheadaouib, A. Hernández-Núñeza
a Servicio de Dermatología, Hospital Universitario de Fuenlabrada, Fuenlabrada, Madrid, Spain
b Servicio de Anatomía Patológica, Hospital Universitario de Fuenlabrada, Fuenlabrada, Madrid, Spain
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    "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Medical History</span><p id="par0005" class="elsevierStylePara elsevierViewall">A 9-year-old boy with a past history of atopic dermatitis and attention deficit and hyperactivity disorder&#44; who was undergoing treatment with methylphenidate and risperidone&#44; visited our department with asymptomatic lesions in the oral cavity that had appeared 7 months earlier&#59; the lesions had been treated with topical antifungal agents &#40;ketoconazole and nystatin&#41;&#44; with no improvement&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Physical Examination</span><p id="par0010" class="elsevierStylePara elsevierViewall">Thick&#44; rough&#44; white plaques that did not detach on scraping were found in the oral cavity&#44; involving the mucosa of both cheeks&#44; the floor of the mouth&#44; and the lateral edges and ventral region of the tongue &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; The rest of the physical examination was normal&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Histopathology</span><p id="par0015" class="elsevierStylePara elsevierViewall">An incisional biopsy of the buccal mucosa was taken&#46; Staining with hematoxylin&#8211;eosin revealed a squamous mucosa with acanthosis and clear cells corresponding to keratinocytes with abundant clear cytoplasm and a small nucleus and foci of a perinuclear cytoplasmic eosinophilic material&#46; The cell wall showed no inflammatory infiltrate &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; PAS staining revealed no mycotic structures&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Other Tests</span><p id="par0020" class="elsevierStylePara elsevierViewall">Mycology culture was negative&#46; Blood tests revealed elevated urea &#40;356&#8239;mg&#47;dL&#41;&#44; and other biochemistry parameters&#44; blood count&#44; complement&#44; and immunoglobulin levels were normal&#46; The serologic study &#40;EBV&#44; <span class="elsevierStyleItalic">Mycoplasma pneumoniae</span>&#44; parvovirus B19&#44; CMV&#41; showed findings compatible with past infection &#40;IgG positive&#41; by EBV and Parvovirus B19&#46;</p><p id="par0025" class="elsevierStylePara elsevierViewall">&#91;&#91;&#63;&#93;&#93;What is your diagnosis&#63;</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Diagnosis</span><p id="par0030" class="elsevierStylePara elsevierViewall">White sponge nevus&#46;</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Clinical Course and Treatment</span><p id="par0035" class="elsevierStylePara elsevierViewall">A wait-and-see approach was adopted due to the benign nature of this entity&#46; The patient is currently being studied for high urea levels at a metabolic diseases department&#46;</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Comment</span><p id="par0040" class="elsevierStylePara elsevierViewall">White sponge nevus is a rare&#44; autosomal dominant hereditary genodermatosis with variable penetrance&#44; although sporadic cases have been reported&#46; It was described by Hyde in 1909 with the name leukokeratosis and in 1935&#44; Cannon coined the term <span class="elsevierStyleItalic">white sponge nevus</span>&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">The pathophysiology is not fully understood&#46; Mutations have been identified in the genes that code for keratins 3 and 14&#44; located on chromosomes 12q13 and 17q21&#8208;q22&#44; respectively&#46; These keratins are expressed specifically in the buccal&#44; anogenital&#44; nasal&#44; esophageal&#44; and laryngeal mucosa&#44; and in other nonkeratinized stratified squamous epithelia&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;4</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Lesions are asymptomatic in most cases and tend to involve the buccal mucosa bilaterally and symmetrically&#44; in the form of white plaques with a rough&#44; spongy appearance&#44; which do not detach on scraping&#46; Rare cases of isolated involvement of the extraoral mucosa have been reported&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> They may be congenital or appear during childhood&#44; as in the case of our patient&#46; They sometimes appear during adolescence and show no preference for sex&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3&#44;5&#44;6</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Histopathology is nonspecific&#46; Of note is the epithelial thickening with parakeratotic hyperkeratosis&#44; acanthosis&#44; and spongiosis&#44; with vacuolization of supra-epidermal keratinocytes and with perinuclear eosinophilic aggregates that correspond to anomalous groupings of keratin tonofilaments&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Diagnosis is based on clinical signs and symptoms and on histopathology findings&#46; A genetic study may also be performed&#46;</p><p id="par0065" class="elsevierStylePara elsevierViewall">Prognosis is good&#46; Cases have been reported in the literature with subsequent development of dysplasia and even epidermoid carcinoma&#44; although the link has not been clearly established&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;5</span></a></p><p id="par0070" class="elsevierStylePara elsevierViewall">Treatment is not required&#44; as the lesions are asymptomatic&#46; Isolated cases of response to chlorhexidine&#44; topical retinoids and topical tetracycline&#44; and oral penicillin have been published&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2&#44;6</span></a></p><p id="par0075" class="elsevierStylePara elsevierViewall">The differential diagnosis must be carried out with other diseases that involve white lesions in the buccal mucosa&#44; such as genodermatosis &#40;congenital pachyonychia&#44; hereditary benign intraepithelial dyskeratosis&#44; and congenital dyskeratosis&#41;&#44; infections &#40;oral candidiasis in its pseudomembranous form&#44; focal epithelial hyperplasia&#44; and syphilis&#41;&#44; inflammatory diseases &#40;lichen planus&#41;&#44; premalignant and malignant lesions &#40;leukoplasia&#44; epidermoid carcinoma&#41;&#44; and other entities such as leukoedema and friction hyperkeratosis&#46;</p><p id="par0080" class="elsevierStylePara elsevierViewall">We report a rare&#44; extensive case of pediatric-onset white sponge nevus&#46; 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