Case and Research Letter
Trauma-induced Adult Xanthogranuloma Mimicking Keloid on the Left Nasal Ala
Xantogranuloma del adulto inducido por traumatismo, que remeda queloide en ala nasal izquierda
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"apellidos" => "Neves Souza" "email" => array:1 [ 0 => "neves.emilly@hotmail.com" ] "referencia" => array:3 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 2 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">*</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "M." "apellidos" => "Rodrigues Batalha" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 2 => array:3 [ "nombre" => "L." "apellidos" => "Horta da Silva" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] 3 => array:3 [ "nombre" => "M." "apellidos" => "Neves Souza" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">d</span>" "identificador" => "aff0020" ] ] ] ] "afiliaciones" => array:4 [ 0 => array:3 [ "entidad" => "Universidade Federal do Espírito Santo (UFES), Vitória, ES, Brazil" "etiqueta" => "a" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Hospital Universitário Cassiano Antônio Moraes (HUCAM), Vitória, ES, Brazil" "etiqueta" => "b" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Intituto Izamar Milidiú da Silva (IMS), Rio de Janeiro, RJ, Brazil" "etiqueta" => "c" "identificador" => "aff0015" ] 3 => array:3 [ "entidad" => "Faculdade Multivix, Vitória - ES, Brazil" "etiqueta" => "d" "identificador" => "aff0020" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Xantogranuloma del adulto inducido por traumatismo, que remeda queloide en ala nasal izquierda" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1124 "Ancho" => 1005 "Tamanyo" => 368084 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">(a) Dense dermal histiocyte infiltrate (HE 100×). (b) Histiocytes and giant multinucleated Touton cells (HE 400×).</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">A 33-year-old healthy female, nurse, Fitzpatrick phototype IV, presented with a history of a piercing proceeded on the left nasal ala in 2019. She referred multiple local traumas in daily activities, such as accidental adhesion of the piercing to clothes or bath towel. In January 2021, a papule appeared on the same nasal region. The patient repeatedly tried to remove the lesion with a small needle, resulting in local bleeding and progressive growth to a tumor. No other lesions or constitutional symptoms have been described.</p><p id="par0010" class="elsevierStylePara elsevierViewall">Dermatological examination revealed small erythematous-brownish nodule, 1.5<span class="elsevierStyleHsp" style=""></span>cm diameter, similar to a keloid adjacent to the nasal piercing (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). No other injuries have been identified. Dermoscopic analysis indicated a yellow-orange central area surrounded by peripheral erythematous border with fine linear telangiectasias, suggestive of xanthogranuloma (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>a and b).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0015" class="elsevierStylePara elsevierViewall">We proceeded the complete excision of the tumor. Histopathological study identified a nodular dermal proliferation with xanthomized histiocytes, foreign body-type multinucleated giant cells and Touton multinucleated cells, along with scattered lymphocytes, plasma cells and eosinophils (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>a and b). Immunohistochemistry appointed positive stains for CD68. S100 and CD1a were absent. BRAF-V600E mutations analysis through real-time polymerase chain reaction (PCR) was negative. Laboratory investigation revealed no abnormalities in routine blood examination parameters, triglyceride levels, liver function or renal function. The tests confirmed the diagnosis of adult xanthogranuloma, and there was clinical improvement after surgical removal. The patient maintains dermatological follow-up with no injury recurrence.</p><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">Xanthogranuloma is the most common non-Langerhans cell histiocytosis.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> It is a benign proliferative disorder, usually described in early childhood as juvenile xanthogranuloma (JXG). Almost 85% of cases of JXG occur in infants less than one year of age, with a male preponderance.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Late-onset lesions in adults is infrequent and represents 10% of all xantogranulomas.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Most reports describe adult xanthogranuloma between the third and fourth decades of life, with equal sex distribution.<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> The pathogenesis is unknown, and the most associated triggers are infections or physical factors,<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> with few studies in the world literature describing trauma-induced xanthogranuloma. The patient described in this report presented an unusual onset of adult xanthogranuloma after recurrent local traumas.</p><p id="par0025" class="elsevierStylePara elsevierViewall">The lesion typically manifests as a reddish or yellowish to brown solitary papulonodule on the head, neck, limbs, and upper trunk.<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> Nasal involvement is even rarer and only three cases on this specific topography have been published.<a class="elsevierStyleCrossRefs" href="#bib0075"><span class="elsevierStyleSup">5–7</span></a> Tan et al. (2014) also reported an unusual presentation of adult xanthogranuloma on the left nasal ala, as we describe herein. However, unlike our case, no trigger factor has been identified.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> A few articles documented multiple lesions and extracutaneous and systemic involvement.<a class="elsevierStyleCrossRefs" href="#bib0060"><span class="elsevierStyleSup">2,4,8</span></a> Dermoscopic evaluation is a powerful tool to define diagnostic hypothesis, revealing the characteristic yellow-orange central area surrounded by a peripheral erythematous border with fine linear telangiectasias.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Differential diagnosis includes dermatofibroma, Langerhans cell histiocytosis, other xanthomatous lesions, mastocytoma, and Spitz nevus.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Diagnostic confirmation is made through skin biopsy. Histopathological analysis identifies dense dermal histiocyte infiltrate and Touton giant cells, which are multinucleated cells, with homogeneous eosinophilic cytoplasmic center and peripheral xanthomatization.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> Immunohistochemistry is also important to define diagnosis and shows positive stains for Factor XIIIa, CD68, CD163, CD14, and fascin. Stains for S100 and CD1a, specific for Langerhans cells, are negative.<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> In cases with atypical histology features or immunohistochemistry suggestive of xanthogranuloma and Langerhans cell histiocytosis (LCH), the investigation of BRAF-V600E mutations is recommended. BRAF-V600E mutations have been described in LCH and can identify LCH coexisting with xanthogranuloma.<a class="elsevierStyleCrossRef" href="#bib0100"><span class="elsevierStyleSup">10</span></a> In this case report, this oncogenic mutation was absent, reaffirming the xanthogranulomatous nature of the tumor.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Usually, no laboratory abnormalities are identified in patients with solitary AXG. However, clinicians should be aware of systemic involvement and hematological malignancies in adults with multiple xantogranulomas.<a class="elsevierStyleCrossRef" href="#bib0090"><span class="elsevierStyleSup">8</span></a> In these cases, laboratory and imaging investigation are essential. AXG generally follows a benign course, with spontaneous regression in approximately 3–6 years.<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Surgical excision, CO<span class="elsevierStyleInf">2</span> laser, and intralesional steroids can be performed, mainly for cosmetic reasons.<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">In this report, the patient's history and clinical examination suggested a keloid lesion. However, dermoscopic analysis revealed typical findings of xanthogranuloma and guided our Dermatology team to an accurate diagnosis. In conclusion, the correct use of the dermatoscope is effective not only to evaluate melanocytic lesions, but also to non-melanocytic, as in the present case. Dermatologists should use this tool daily in every physical examination to improve diagnostic accuracy. After directing the diagnostic hypothesis, the confirmation was established through histopathology and immunohistochemistry, which were also compatible with adult xanthogranuloma.</p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Conflict of interests</span><p id="par0045" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflict of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:2 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Conflict of interests" ] 1 => array:1 [ "titulo" => "References" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "multimedia" => array:3 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 919 "Ancho" => 1074 "Tamanyo" => 99394 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Small erythematous-brownish nodule adjacent to the nasal piercing.</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" => 419 "Ancho" => 1005 "Tamanyo" => 63030 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">(a and b) Dermoscopic analysis: yellow-orange central area surrounded by peripheral erythematous border with fine linear telangiectasias.</p>" ] ] 2 => array:7 [ "identificador" => "fig0015" "etiqueta" => "Figure 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 1124 "Ancho" => 1005 "Tamanyo" => 368084 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">(a) Dense dermal histiocyte infiltrate (HE 100×). (b) Histiocytes and giant multinucleated Touton cells (HE 400×).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0055" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Juvenile xanthogranuloma: an entity with a wide clinical spectrum" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:3 [ 0 => "M.J. Hernández-San Martín" 1 => "P. Vargas-Mora" 2 => "L. 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