Casos para el diagnóstico
Erythematous nodule on the upper lip
Nódulo eritematoso en labio superior
M.D. Pegalajar-García, I. Pérez-López, F.J. Navarro-Triviño
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Servicio de Dermatología, Hospital Universitario San Cecilio, Granada, Spain
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Células endoteliales prominentes con abundante citoplasma eosinófilo (H&E x10) (B). Células endoteliales prominentes con abundante citoplasma eosinófilo rodeadas principalmente de linfocitos y algunos eosinófilos aislados (H&E x20) (C) Resolución completa de los nódulos eritematosos tras tratamiento con láser.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "M.D. Pegalajar-García, I. Pérez-López, F.J. Navarro-Triviño" "autores" => array:3 [ 0 => array:2 [ "nombre" => "M.D." "apellidos" => "Pegalajar-García" ] 1 => array:2 [ "nombre" => "I." "apellidos" => "Pérez-López" ] 2 => array:2 [ "nombre" => "F.J." 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"apellidos" => "Navarro-Triviño" "email" => array:1 [ 0 => "fntmed@gmail.com" ] "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Servicio de Dermatología, Hospital Universitario San Cecilio, Granada, Spain" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Nódulo eritematoso en labio superior" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 621 "Ancho" => 930 "Tamanyo" => 213735 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Intradermal proliferation of lobulated vascular structures with prominent endothelium and inflammatory infiltrate (panoramic Hematoxylin & Eosin) (A). Prominent endothelial cells with abundant eosinophilic cytoplasm (H&E x10) (B). Prominent endothelial cells with abundant eosinophilic cytoplasm mainly surrounded by lymphocytes and a few isolated eosinophils (H&E x20) (C). Complete resolution of erythematous nodules after laser treatment.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Clinical history</span><p id="par0005" class="elsevierStylePara elsevierViewall">A 37-year-old man, with no personal or family history of interest presented with a 2-month history of asymptomatic lesions on his upper lip. He denied the occurrence of any hemorrhages and had not seen any ulcers in the lesions.</p><p id="par0010" class="elsevierStylePara elsevierViewall">Physical examination revealed the presence of an erythematous nodule of a smooth surface and semi-solid consistency on the upper lip (<a class="elsevierStyleCrossRef" href="#fig0005">fig. 1</a>A), along with an additional asymptomatic papular lesion on the same mucosa (<a class="elsevierStyleCrossRef" href="#fig0005">fig. 1</a>B). Dermoscopy (<a class="elsevierStyleCrossRef" href="#fig0005">fig. 1</a>C) revealed the presence of a homogeneous red pattern with whitish areas surrounded by dilated tortuous vessels, some resembling a forked vascular pattern.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">Histopathological examination of a 4<span class="elsevierStyleHsp" style=""></span>mm punch biopsy confirmed the presence of an intradermal tumor lesion composed of a vascular proliferation of prominent endothelium, along with a predominantly lymphocytic inflammatory infiltrate with few interspersed eosinophils.</p><p id="par0020" class="elsevierStylePara elsevierViewall">No additional tests were performed for diagnostic purposes.</p><p id="par0025" class="elsevierStylePara elsevierViewall"><span class="elsevierStyleBold">What is the diagnosis?</span></p><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Diagnosis and comments</span><p id="par0030" class="elsevierStylePara elsevierViewall">The diagnosis achieved was angiolymphoid hyperplasia with eosinophilia (ALHE).</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Disease progression and treatment</span><p id="par0035" class="elsevierStylePara elsevierViewall">Although the patient was informed about the possibility of spontaneous lesion resolution, he insisted on being treated, and received 2 sessions (595<span class="elsevierStyleHsp" style=""></span>nm, 7<span class="elsevierStyleHsp" style=""></span>mm diameter, 7<span class="elsevierStyleHsp" style=""></span>J/cm<span class="elsevierStyleSup">2</span> fluence, and 0.5<span class="elsevierStyleHsp" style=""></span>ms pulses; 2 pulses per lesion; air cooling) of pulsed dye laser (PDL), achieving complete resolution with an almost perfect aesthetic appearance (<a class="elsevierStyleCrossRef" href="#fig0010">fig. 2</a>A-C). The skin alterations did not relapse 6 months into treatment (<a class="elsevierStyleCrossRef" href="#fig0010">fig. 2</a>D).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Comments</span><p id="par0040" class="elsevierStylePara elsevierViewall">ALHE is a subtype of epithelioid hemangioma, within benign angiolymphoproliferative processes, which is associated with a greater inflammatory component. It is a rare entity more prevalent in middle-aged women characterized by the appearance of erythematous-violaceous papules and nodules and is usually located on the head and neck. Lip or oral mucosa involvement is a rare finding,<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a> as it predominantly affects the periauricular area (36.3%), face (28.2%), and scalp (17.3%).<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a> Although these papules and nodules often remain asymptomatic, they may be associated with pruritus.<a class="elsevierStyleCrossRef" href="#bib0045"><span class="elsevierStyleSup">2</span></a> Additionally, although their etiopathogenesis is unknown to this date, they could be due to reactive vascular proliferation in response to infections, hormonal factors, or trauma.<a class="elsevierStyleCrossRef" href="#bib0050"><span class="elsevierStyleSup">3</span></a> The occurrence of ALHE after getting a tattoo done supports this latter hypothesis.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">4</span></a> Histological examination shows the proliferation of vascular structures of varying calibers with prominent epithelioid endothelial cells, along with a lymphocytic infiltrate and the presence of eosinophils.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">1</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">Differential diagnosis includes entities such as cutaneous epithelioid angiomatoid nodule (CEAN), cutaneous sarcoidosis, pyogenic granuloma, cutaneous lymphoma, Kaposi's sarcoma, and various types of epithelioid neoplasms. Among these, ALHE should be distinguished from other vascular epithelioid lesions such as epithelioid hemangioendothelioma, pseudomyogenic hemangioendothelioma, and epithelioid angiosarcoma (EA).</p><p id="par0050" class="elsevierStylePara elsevierViewall">Histopathological characteristics and the use of an immunohistochemical panel are used to distinguish one entity from the other. Although ALHE generally expresses the switch regulatory protein 3 FosB,<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">5</span></a> conditions such as pseudomyogenic hemangioendothelioma can also express it. Superficial location, good delimitation, and the absence of an infiltrative pattern with a low degree of nuclear atypia and mitotic figures, and negativity for podoplanin and <span class="elsevierStyleItalic">avian myelocytomatosis viral oncogene homolog</span>, distinguish ALHE from EA.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">5</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">The management of ALHE is not standardized. Excision is usually performed to treat single lesions, while therapies with variable results include topical and intralesional corticosteroids, cryocoagulation, pentoxifylline, or various laser modalities are used to treat multiple lesions.<a class="elsevierStyleCrossRefs" href="#bib0050"><span class="elsevierStyleSup">3,6</span></a> In published cases, PDL<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">7</span></a> has turned out to be a safe therapy that improves symptoms and the aesthetic appearance of lesions, even leading to their disappearance, as it happened with our case. The mechanism of action of PDL induces photothermolysis, which eventually leads to the selective destruction of vascular lesions, thus reducing damage to surrounding structures, and facilitating faster healing compared with other therapies such as carbon dioxide.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">7</span></a> This has turned PDL into an attractive option for the management of ALHE since this is a benign condition that tends to recur frequently despite previous therapies.</p></span></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Funding</span><p id="par0060" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Conflicts of interest</span><p id="par0065" class="elsevierStylePara elsevierViewall">None declared.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:4 [ 0 => array:3 [ "identificador" => "sec0005" "titulo" => "Clinical history" "secciones" => array:3 [ 0 => array:2 [ "identificador" => "sec0010" "titulo" => "Diagnosis and comments" ] 1 => array:2 [ "identificador" => "sec0015" "titulo" => "Disease progression and treatment" ] 2 => array:2 [ "identificador" => "sec0020" "titulo" => "Comments" ] ] ] 1 => array:2 [ "identificador" => "sec0025" "titulo" => "Funding" ] 2 => array:2 [ "identificador" => "sec0030" "titulo" => "Conflicts of interest" ] 3 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 526 "Ancho" => 930 "Tamanyo" => 121260 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Smooth-surfaced erythematous nodule located on the upper lip of angiomatous appearance (A), erythematous papule in the upper lip internal mucosa (black arrow) (B). Homogeneous erythematous pattern with whitish areas and dilated tortuous vessels (Dermlite® DL4, Dermlite LLC, United States) (C).</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 621 "Ancho" => 930 "Tamanyo" => 213735 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Intradermal proliferation of lobulated vascular structures with prominent endothelium and inflammatory infiltrate (panoramic Hematoxylin & Eosin) (A). Prominent endothelial cells with abundant eosinophilic cytoplasm (H&E x10) (B). Prominent endothelial cells with abundant eosinophilic cytoplasm mainly surrounded by lymphocytes and a few isolated eosinophils (H&E x20) (C). Complete resolution of erythematous nodules after laser treatment.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:7 [ 0 => array:3 [ "identificador" => "bib0040" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Angiolymphoid hyperplasia with eosinophilia on lip in a 10-year-old girl" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "H.T. Chaturvedi" 1 => "C. Chaturvedi" 2 => "S. Thammaiah" ] ] ] ] ] "host" => array:1 [ 0 => array:2 [ "doi" => "10.4103/jomfp.jomfp_447_21" "Revista" => array:6 [ "tituloSerie" => "J Oral Maxillofac Pathol." 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