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with a 20&#37; of ki67&#44; and a positive in situ hybridization for Epstein-Barr virus &#40;EBER&#41; test &#40;<a class="elsevierStyleCrossRefs" href="#fig0010">Figs&#46; 2 and 3</a>&#41;&#46; A conjunctiva biopsy was performed&#44; with evidence of necrotic tissue&#44; and a positive polymerase chain reaction for EBV&#46; Viral load for EBV in blood was positive &#40;197&#44;929 copies&#47;mL&#41;&#46; With all of the above&#44; a diagnosis of HV-LPD was performed&#46; CT scans report cervical adenopathies and hepatosplenomegaly&#46; Biopsy of cervical node and bone marrow was negative for malignancy&#46; Proper treatment with oral thalidomide 100&#8239;mg QD was initiated&#44; achieving clinical response&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">American cases of HV-LPD have been described in children of Mexico&#44; Peru&#44; and Bolivia&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> According to some studies viral DNA is elevated in most patients&#44; suggesting a chronic EBV infection and a genetic susceptibility for defective EBV-specific immunity&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;4</span></a> Clinically&#44; they present as papulovesicular eruptions&#44; with necrotic centers&#44; in sun-exposed and non-exposed areas&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Lie et al&#46; report 12&#37; of patients with ocular symptoms&#44; including corneal nebula&#44; conjunctival swelling&#44; photophobia&#44; and tearing&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Unfortunately&#44; our patient had severe ocular involvement which&#44; in the best of our knowledge&#44; there are no previous reports on such association&#46; Therefore&#44; we propose this new feature&#44; due to the clinical picture and positivity for EBV DNA&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">Histologically&#44; the epidermis displays extensive ulceration with necrosis and angiocentricity as a common finding in the vessels&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Frequent findings include a dense infiltrate of small-to middle size atypical lymphoid cell&#44; pleomorphic nuclei&#44; mainly located around adnexae and blood vessels&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;4</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Immunohistochemistry reported a lymphoid population &#40;CD3<span class="elsevierStyleSup">&#43;</span>&#44; CD5<span class="elsevierStyleSup">&#43;</span>&#44; and CD7<span class="elsevierStyleSup">&#43;</span>&#41;<span class="elsevierStyleSup">1</span> with cytotoxic or natural killer phenotype &#40;CD8<span class="elsevierStyleSup">&#43;</span> and CD56<span class="elsevierStyleSup">&#43;</span> respectively&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The most frequent phenotype is a cytotoxic lymphoid infiltrate&#44; like in our patient&#44; with positive cytotoxic markers&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;3</span></a> Performance of EBER&#8217;s is encoded for the detection of EBV RNA&#44; positivity of this test have been report up to 100&#37;&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3&#44;5</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The main differential diagnoses are classic hydroa vacciniforme&#46; As HV-LPD&#44; classic hydroa vacciniforme appears in children&#44; has a papulovesicular eruption with a crusted center&#44; and posterior varioliforme scarring&#46; Nonetheless&#44; its localization is limited to sun-exposed areas&#44; with no facial edema nor systemic compromise&#46; Histologic differential diagnosis includes other lymphomas associated with EBV&#46; For example&#44; nasal NK lymphoma is characterized by a rapidly progressive clinical course&#44; positive CD56 and negative CD8 on the immunohistochemistry&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">There are no treatment guidelines due to the rarity of the disease&#46; Beltran et al&#46; consider thalidomide as a useful treatment&#44; due to its anti-inflammatory and antiproliferative properties&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Four Peruvian patients received thalidomide 100&#8239;mg orally daily with different results&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> We report a 12-month clinical remission in our patient&#44; following thalidomide treatment&#44; supporting the use of it as a first-line immunomodulating agent&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0035" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Authors&#8217; contributions</span><p id="par0040" class="elsevierStylePara elsevierViewall">Juliana Ordo&#241;ez-Parra&#58; Patient assessment&#59; literature review&#59; manuscript creation&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Maddy Mej&#237;a Cortes&#58; Patient assessment&#59; literature review&#59; manuscript creation&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Margarita Tamayo-Buend&#237;a&#58; Patient assessment&#59; literature review&#59; manuscript creation&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">Ana Mar&#237;a Infante G&#243;mez&#58; Patient assessment and literature review&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0060" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span></span>"
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Case Letter
Hydroa vacciniforme-like lymphoproliferative disorder (HV-LPD) is an Epstein-Barr virus (EBV) associated disease
Juliana Ordoñez-Parra
Autor para correspondência
juliana.ordonez19@gmail.com

Corresponding author.
, Maddy Mejía Cortes, Maria Margarita Tamayo-Buendía, Ana María Infante Gómez
Hospital Universitario San Ignacio, Bogotá, Colombia
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    "titulo" => "Hydroa vacciniforme-like lymphoproliferative disorder &#40;HV-LPD&#41; is an Epstein-Barr virus &#40;EBV&#41; associated disease"
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          "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Positive in-situ hybridization for EBV &#40;black arrows&#41; &#40;Hematoxylin &#38; eosin&#44; &#215;100&#41;&#46;</p>"
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We present a 12-year-old Hispanic male with a 6-year history of &#8220;nodules&#8221; that ulcerated in the face&#44; lower and upper limbs which left multiple scars&#46; He attended for 20-days of facial edema&#44; associated with a decrease in visual acuity&#46; The ophthalmologist reported necrosis of the left eye&#46; During the physical examination he presented periorbital edema&#44; left frontal vesico-blisters&#44; which left varioliform scars &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46; A skin biopsy was performed with evidence of epidermal necrosis&#44; atypical perivascular&#44; and perianexial lymphoid infiltrates with angiocentricity&#46; Immunohistochemistry was compatible with cytotoxic T lymphocytes &#40;CD3<span class="elsevierStyleSup">&#43;</span> CD8<span class="elsevierStyleSup">&#43;</span> Perforine<span class="elsevierStyleSup">&#43;</span> CD56-&#41; with a 20&#37; of ki67&#44; and a positive in situ hybridization for Epstein-Barr virus &#40;EBER&#41; test &#40;<a class="elsevierStyleCrossRefs" href="#fig0010">Figs&#46; 2 and 3</a>&#41;&#46; A conjunctiva biopsy was performed&#44; with evidence of necrotic tissue&#44; and a positive polymerase chain reaction for EBV&#46; Viral load for EBV in blood was positive &#40;197&#44;929 copies&#47;mL&#41;&#46; With all of the above&#44; a diagnosis of HV-LPD was performed&#46; CT scans report cervical adenopathies and hepatosplenomegaly&#46; Biopsy of cervical node and bone marrow was negative for malignancy&#46; Proper treatment with oral thalidomide 100&#8239;mg QD was initiated&#44; achieving clinical response&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">American cases of HV-LPD have been described in children of Mexico&#44; Peru&#44; and Bolivia&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a> According to some studies viral DNA is elevated in most patients&#44; suggesting a chronic EBV infection and a genetic susceptibility for defective EBV-specific immunity&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#44;4</span></a> Clinically&#44; they present as papulovesicular eruptions&#44; with necrotic centers&#44; in sun-exposed and non-exposed areas&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Lie et al&#46; report 12&#37; of patients with ocular symptoms&#44; including corneal nebula&#44; conjunctival swelling&#44; photophobia&#44; and tearing&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Unfortunately&#44; our patient had severe ocular involvement which&#44; in the best of our knowledge&#44; there are no previous reports on such association&#46; Therefore&#44; we propose this new feature&#44; due to the clinical picture and positivity for EBV DNA&#46;</p><p id="par0015" class="elsevierStylePara elsevierViewall">Histologically&#44; the epidermis displays extensive ulceration with necrosis and angiocentricity as a common finding in the vessels&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Frequent findings include a dense infiltrate of small-to middle size atypical lymphoid cell&#44; pleomorphic nuclei&#44; mainly located around adnexae and blood vessels&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;4</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Immunohistochemistry reported a lymphoid population &#40;CD3<span class="elsevierStyleSup">&#43;</span>&#44; CD5<span class="elsevierStyleSup">&#43;</span>&#44; and CD7<span class="elsevierStyleSup">&#43;</span>&#41;<span class="elsevierStyleSup">1</span> with cytotoxic or natural killer phenotype &#40;CD8<span class="elsevierStyleSup">&#43;</span> and CD56<span class="elsevierStyleSup">&#43;</span> respectively&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The most frequent phenotype is a cytotoxic lymphoid infiltrate&#44; like in our patient&#44; with positive cytotoxic markers&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#8211;3</span></a> Performance of EBER&#8217;s is encoded for the detection of EBV RNA&#44; positivity of this test have been report up to 100&#37;&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;3&#44;5</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The main differential diagnoses are classic hydroa vacciniforme&#46; As HV-LPD&#44; classic hydroa vacciniforme appears in children&#44; has a papulovesicular eruption with a crusted center&#44; and posterior varioliforme scarring&#46; Nonetheless&#44; its localization is limited to sun-exposed areas&#44; with no facial edema nor systemic compromise&#46; Histologic differential diagnosis includes other lymphomas associated with EBV&#46; For example&#44; nasal NK lymphoma is characterized by a rapidly progressive clinical course&#44; positive CD56 and negative CD8 on the immunohistochemistry&#46;</p><p id="par0030" class="elsevierStylePara elsevierViewall">There are no treatment guidelines due to the rarity of the disease&#46; Beltran et al&#46; consider thalidomide as a useful treatment&#44; due to its anti-inflammatory and antiproliferative properties&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Four Peruvian patients received thalidomide 100&#8239;mg orally daily with different results&#46;<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> We report a 12-month clinical remission in our patient&#44; following thalidomide treatment&#44; supporting the use of it as a first-line immunomodulating agent&#46;</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0035" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Authors&#8217; contributions</span><p id="par0040" class="elsevierStylePara elsevierViewall">Juliana Ordo&#241;ez-Parra&#58; Patient assessment&#59; literature review&#59; manuscript creation&#46;</p><p id="par0045" class="elsevierStylePara elsevierViewall">Maddy Mej&#237;a Cortes&#58; Patient assessment&#59; literature review&#59; manuscript creation&#46;</p><p id="par0050" class="elsevierStylePara elsevierViewall">Margarita Tamayo-Buend&#237;a&#58; Patient assessment&#59; literature review&#59; manuscript creation&#46;</p><p id="par0055" class="elsevierStylePara elsevierViewall">Ana Mar&#237;a Infante G&#243;mez&#58; Patient assessment and literature review&#46;</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0060" class="elsevierStylePara elsevierViewall">None declared&#46;</p></span></span>"
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Anais Brasileiros de Dermatologia (Portuguese)
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