Case Letter
Hydroa vacciniforme-like lymphoproliferative disorder (HV-LPD) is an Epstein-Barr virus (EBV) associated disease
Juliana Ordoñez-Parra
, Maddy Mejía Cortes, Maria Margarita Tamayo-Buendía, Ana María Infante Gómez
Corresponding author
Hospital Universitario San Ignacio, Bogotá, Colombia
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"autores" => array:1 [ 0 => array:4 [ "autoresLista" => "Juliana Ordoñez-Parra, Maddy Mejía Cortes, Maria Margarita Tamayo-Buendía, Ana María Infante Gómez" "autores" => array:4 [ 0 => array:4 [ "nombre" => "Juliana" "apellidos" => "Ordoñez-Parra" "email" => array:1 [ 0 => "juliana.ordonez19@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "*" "identificador" => "cor0005" ] ] ] 1 => array:2 [ "nombre" => "Maddy" "apellidos" => "Mejía Cortes" ] 2 => array:2 [ "nombre" => "Maria Margarita" "apellidos" => "Tamayo-Buendía" ] 3 => array:2 [ "nombre" => "Ana María" "apellidos" => "Infante Gómez" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Hospital Universitario San Ignacio, Bogotá, Colombia" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 538 "Ancho" => 750 "Tamanyo" => 96472 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Positive in-situ hybridization for EBV (black arrows) (Hematoxylin & eosin, ×100).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">We present a 12-year-old Hispanic male with a 6-year history of “nodules” that ulcerated in the face, lower and upper limbs which left multiple scars. He attended for 20-days of facial edema, associated with a decrease in visual acuity. The ophthalmologist reported necrosis of the left eye. During the physical examination he presented periorbital edema, left frontal vesico-blisters, which left varioliform scars (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). A skin biopsy was performed with evidence of epidermal necrosis, atypical perivascular, and perianexial lymphoid infiltrates with angiocentricity. Immunohistochemistry was compatible with cytotoxic T lymphocytes (CD3<span class="elsevierStyleSup">+</span> CD8<span class="elsevierStyleSup">+</span> Perforine<span class="elsevierStyleSup">+</span> CD56-) with a 20% of ki67, and a positive in situ hybridization for Epstein-Barr virus (EBER) test (<a class="elsevierStyleCrossRefs" href="#fig0010">Figs. 2 and 3</a>). A conjunctiva biopsy was performed, with evidence of necrotic tissue, and a positive polymerase chain reaction for EBV. Viral load for EBV in blood was positive (197,929 copies/mL). With all of the above, a diagnosis of HV-LPD was performed. CT scans report cervical adenopathies and hepatosplenomegaly. Biopsy of cervical node and bone marrow was negative for malignancy. Proper treatment with oral thalidomide 100 mg QD was initiated, achieving clinical response.</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, Peru, and Bolivia.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,2</span></a> According to some studies viral DNA is elevated in most patients, suggesting a chronic EBV infection and a genetic susceptibility for defective EBV-specific immunity.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,4</span></a> Clinically, they present as papulovesicular eruptions, with necrotic centers, in sun-exposed and non-exposed areas.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> Lie et al. report 12% of patients with ocular symptoms, including corneal nebula, conjunctival swelling, photophobia, and tearing.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Unfortunately, our patient had severe ocular involvement which, in the best of our knowledge, there are no previous reports on such association. Therefore, we propose this new feature, due to the clinical picture and positivity for EBV DNA.</p><p id="par0015" class="elsevierStylePara elsevierViewall">Histologically, the epidermis displays extensive ulceration with necrosis and angiocentricity as a common finding in the vessels.<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, pleomorphic nuclei, mainly located around adnexae and blood vessels.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,4</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">Immunohistochemistry reported a lymphoid population (CD3<span class="elsevierStyleSup">+</span>, CD5<span class="elsevierStyleSup">+</span>, and CD7<span class="elsevierStyleSup">+</span>)<span class="elsevierStyleSup">1</span> with cytotoxic or natural killer phenotype (CD8<span class="elsevierStyleSup">+</span> and CD56<span class="elsevierStyleSup">+</span> respectively).<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The most frequent phenotype is a cytotoxic lymphoid infiltrate, like in our patient, with positive cytotoxic markers.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1–3</span></a> Performance of EBER’s is encoded for the detection of EBV RNA, positivity of this test have been report up to 100%.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,3,5</span></a></p><p id="par0025" class="elsevierStylePara elsevierViewall">The main differential diagnoses are classic hydroa vacciniforme. As HV-LPD, classic hydroa vacciniforme appears in children, has a papulovesicular eruption with a crusted center, and posterior varioliforme scarring. Nonetheless, its localization is limited to sun-exposed areas, with no facial edema nor systemic compromise. Histologic differential diagnosis includes other lymphomas associated with EBV. For example, nasal NK lymphoma is characterized by a rapidly progressive clinical course, positive CD56 and negative CD8 on the immunohistochemistry.</p><p id="par0030" class="elsevierStylePara elsevierViewall">There are no treatment guidelines due to the rarity of the disease. Beltran et al. consider thalidomide as a useful treatment, due to its anti-inflammatory and antiproliferative properties.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> Four Peruvian patients received thalidomide 100 mg orally daily with different results.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> We report a 12-month clinical remission in our patient, following thalidomide treatment, supporting the use of it as a first-line immunomodulating agent.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0035" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Authors’ contributions</span><p id="par0040" class="elsevierStylePara elsevierViewall">Juliana Ordoñez-Parra: Patient assessment; literature review; manuscript creation.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Maddy Mejía Cortes: Patient assessment; literature review; manuscript creation.</p><p id="par0050" class="elsevierStylePara elsevierViewall">Margarita Tamayo-Buendía: Patient assessment; literature review; manuscript creation.</p><p id="par0055" class="elsevierStylePara elsevierViewall">Ana María Infante Gómez: Patient assessment and literature review.</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.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Financial support" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Authors’ contributions" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Conflicts of interest" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "fechaRecibido" => "2020-04-29" "fechaAceptado" => "2020-06-21" "NotaPie" => array:2 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">How to cite this article: Ordoñez-Parra J, Mejía Cortes M, Tamayo-Buendía MM, Infante Gómez AM. Hydroa vacciniforme-like lymphoproliferative disorder (HV-LPD) is an Epstein-Barr virus (EBV) associated disease. An Bras Dermatol. 2021;96:388–90.</p>" ] 1 => array:2 [ "etiqueta" => "☆☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Study conducted at the Hospital Universitario San Ignacio, Bogotá, Colombia.</p>" ] ] "multimedia" => array:3 [ 0 => array:8 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 633 "Ancho" => 900 "Tamanyo" => 54047 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">(A), Periorbital edema, and erythema with left frontal vesico-blisters with hemorrhagic content. (B), Multiple atrophic and some anetodermic scars on the lower limbs.</p>" ] ] 1 => array:8 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 536 "Ancho" => 1500 "Tamanyo" => 130556 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">(A), Atypical perivascular and perianexial lymphoid infiltrates. (B), Angiocentricity.</p>" ] ] 2 => array:8 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 538 "Ancho" => 750 "Tamanyo" => 96472 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Positive in-situ hybridization for EBV (black arrows) (Hematoxylin & eosin, ×100).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:5 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Clinicopathologic Features of Hydroa Vacciniforme – Like Lymphoma: A Series of 9 Patients" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "M. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 15 | 8 | 23 |
| 2024 October | 105 | 75 | 180 |
| 2024 September | 137 | 105 | 242 |
| 2024 August | 153 | 143 | 296 |
| 2024 July | 117 | 108 | 225 |
| 2024 June | 109 | 68 | 177 |
| 2024 May | 85 | 51 | 136 |
| 2024 April | 94 | 80 | 174 |
| 2024 March | 104 | 74 | 178 |
| 2024 February | 93 | 80 | 173 |
| 2024 January | 76 | 46 | 122 |
| 2023 December | 56 | 59 | 115 |
| 2023 November | 78 | 77 | 155 |
| 2023 October | 68 | 76 | 144 |
| 2023 September | 72 | 75 | 147 |
| 2023 August | 72 | 46 | 118 |
| 2023 July | 170 | 34 | 204 |
| 2023 June | 86 | 46 | 132 |
| 2023 May | 83 | 17 | 100 |
| 2023 April | 70 | 23 | 93 |
| 2023 March | 76 | 45 | 121 |
| 2023 February | 64 | 41 | 105 |
| 2023 January | 63 | 42 | 105 |
| 2022 December | 67 | 33 | 100 |
| 2022 November | 93 | 58 | 151 |
| 2022 October | 96 | 53 | 149 |
| 2022 September | 50 | 62 | 112 |
| 2022 August | 75 | 48 | 123 |
| 2022 July | 78 | 61 | 139 |
| 2022 June | 59 | 56 | 115 |
| 2022 May | 39 | 41 | 80 |
| 2022 April | 56 | 65 | 121 |
| 2022 March | 56 | 66 | 122 |
| 2022 February | 30 | 42 | 72 |
| 2022 January | 49 | 74 | 123 |
| 2021 December | 43 | 62 | 105 |
| 2021 November | 57 | 71 | 128 |
| 2021 October | 72 | 95 | 167 |
| 2021 September | 50 | 49 | 99 |
| 2021 August | 64 | 70 | 134 |
| 2021 July | 66 | 58 | 124 |
| 2021 June | 109 | 109 | 218 |
| 2021 May | 109 | 99 | 208 |
| 2021 April | 145 | 275 | 420 |
| 2021 March | 1 | 3 | 4 |
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