Case Letter
Epithelioid sarcoma of the upper limb with nine years of evolution
Maraya de Jesus Semblano Bittencourt, Caren dos Santos Lima, Aline de Lima Dias
, Camilla Côrrea Neri
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Department of Dermatology, Centro Universitário do Estado do Pará, Belém, PA, Brazil
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The etiology remains unknown, with a high percentage of lymph node and lung metastasis.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> The authors report a case of a 49-year-old male patient, who presented multiple painful ulcers on the left upper limb for nine years, which increased in number and extension and did not heal. The condition started with a left wrist injury after blunt trauma with a hammer. Upon dermatological examination, the authors observed the presence of multiple ulcers with bloody, elevated, infiltrated edges, some with a purulent floor, others with a granular and bloody floor, grouped, of varying sizes, affecting the entire extension of the left upper correct to CC BY limb (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>). There were also nodules, one of them soft and erythematous-violaceous, and others with exulcerated surfaces, in a linear distribution on the left shoulder. He presented weight loss of 10 kg in the last year. The patient also had severe and continuous pain, requiring frequent use of analgesics and anti-inflammatory drugs. He had already undergone several oral, parenteral, and local antibiotic therapies, without improvement. The initial hypotheses were mycobacteriosis and deep mycoses. Chest radiography showed linear dense areas in the pulmonary bases and a small nodular image projected in the left lung field measuring approximately 0.3 cm. Histopathological examination showed an infiltrative neoplasm consisting of epithelioid and spindle cells with eosinophilic cytoplasm and irregular nuclei with evident nucleoli, fibrosis of the adjacent tissue, and focal necrosis (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). The immunohistochemical examination showed expression for cytokeratins AE1/AE3, CD34, and complete loss of INI-1 expression. These findings are consistent with the diagnosis of ES. The ES initially presents as painless, localized, slowly growing nodules, which evolve to chronic ulcers that do not heal, with raised margins, usually in the distal limbs of young adults, although there are reports of cases affecting children and the elderly. Despite its slow growth, it can be an extremely aggressive tumor with a clinical course characterized by high rates of local recurrence and metastatic potential, especially for lymph nodes and lungs.<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3,4</span></a> Histopathologically, it presents a nodular arrangement of epithelioid neoplastic cells with central degeneration and necrosis. Vascular invasion is rare. Neoplastic cells are oval or polygonal and large, similar to rhabdomyosarcoma. The spindle cells resemble fibrosarcoma or malignant fibrohistiocytoma.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> In immunohistochemical analysis, ES characteristically shows reactivity for epithelial markers, as well as for mesenchymal markers. It is consistently positive for cytokeratin, EMA, and vimentin expression. There is a positive reaction with CD34 in almost 50% of cases. INI-1 is deficient in approximately 90% of cases.<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1,4</span></a> Complete surgical resection is the basis of curative therapy for ES. Some authors strongly recommend radical local excision, with amputation, as the first operative procedure.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> The authors report a rare case of upper limb ES, with long evolution before the diagnosis; attention is drawn to the important role of the dermatologist in the clinical recognition of this neoplasm.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Financial support</span><p id="par0010" class="elsevierStylePara elsevierViewall">None declared.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Authors’ contributions</span><p id="par0015" class="elsevierStylePara elsevierViewall">Maraya de Jesus Semblano Bittencourt: Approval of the final version of the manuscript; collection, analysis, and interpretation of data; critical review of the literature; critical review of the manuscript.</p><p id="par0020" class="elsevierStylePara elsevierViewall">Caren dos Santos Lima: Drafting and editing of the manuscript; collection, analysis, and interpretation of data; critical review of the literature; critical review of the manuscript.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Aline de Lima Days: Approval of the final version of the manuscript; Drafting and editing of the manuscript; intellectual participation in propaedeutic and/or therapeutic conduct of studied cases; critical review of the literature; critical review of the manuscript.</p><p id="par0030" class="elsevierStylePara elsevierViewall">Camilla Côrrea Neri: Drafting and editing of the manuscript; collection, analysis, and interpretation of data; critical review of the literature; critical review of the manuscript.</p></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Conflicts of interest</span><p id="par0035" 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" => "2020-01-21" "fechaAceptado" => "2020-06-10" "NotaPie" => array:2 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">How to cite this article: Bittencourt MJS, Lima CS, Dias AL, Neri CC. Epithelioid sarcoma of the upper limb with nine years of evolution. An Bras Dermatol. 2021;96:11–7.</p>" ] 1 => array:2 [ "etiqueta" => "☆☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0010">Study conducted at the Centro Universitário do Estado do Pará, Belém, PA, Brazil.</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" => 390 "Ancho" => 900 "Tamanyo" => 44551 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0005" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Multiple ulcers with raised and infiltrated edges, some with a purulent floor and others with a granular and bloody floor.</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" => 561 "Ancho" => 900 "Tamanyo" => 68258 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0010" "detalle" => "Figure " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Detail of multiple ulcers with raised and infiltrated edges.</p>" ] ] 2 => array:8 [ "identificador" => "fig0015" "etiqueta" => "Fig. 3" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr3.jpeg" "Alto" => 754 "Ancho" => 750 "Tamanyo" => 205530 ] ] "detalles" => array:1 [ 0 => array:3 [ "identificador" => "at0015" "detalle" => "Fig. " "rol" => "short" ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">Histopathological examination showing an infiltrative neoplasm consisting of epithelioid and spindle cells with eosinophilic cytoplasm and irregular nuclei with evident nucleoli (Hematoxylin & eosin, ×400).</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" => "Epithelioid sarcoma: diagnostic features and genetics" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:4 [ 0 => "K. 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| Ano/Mês | Html | Total | |
|---|---|---|---|
| 2024 Novembro | 17 | 9 | 26 |
| 2024 Outubro | 89 | 73 | 162 |
| 2024 Setembro | 136 | 70 | 206 |
| 2024 Agosto | 150 | 105 | 255 |
| 2024 Julho | 147 | 100 | 247 |
| 2024 Junho | 96 | 88 | 184 |
| 2024 Maio | 83 | 62 | 145 |
| 2024 Abril | 94 | 89 | 183 |
| 2024 Março | 76 | 66 | 142 |
| 2024 Fevereiro | 80 | 76 | 156 |
| 2024 Janeiro | 59 | 47 | 106 |
| 2023 Dezembro | 53 | 55 | 108 |
| 2023 Novembro | 61 | 77 | 138 |
| 2023 Outubro | 50 | 71 | 121 |
| 2023 Setembro | 67 | 63 | 130 |
| 2023 Agosto | 66 | 63 | 129 |
| 2023 Julho | 45 | 115 | 160 |
| 2023 Junho | 53 | 38 | 91 |
| 2023 Maio | 47 | 26 | 73 |
| 2023 Abril | 33 | 36 | 69 |
| 2023 Março | 53 | 28 | 81 |
| 2023 Fevereiro | 37 | 34 | 71 |
| 2023 Janeiro | 46 | 61 | 107 |
| 2022 Dezembro | 51 | 42 | 93 |
| 2022 Novembro | 51 | 56 | 107 |
| 2022 Outubro | 66 | 65 | 131 |
| 2022 Setembro | 52 | 115 | 167 |
| 2022 Agosto | 35 | 69 | 104 |
| 2022 Julho | 40 | 56 | 96 |
| 2022 Junho | 40 | 86 | 126 |
| 2022 Maio | 36 | 55 | 91 |
| 2022 Abril | 38 | 53 | 91 |
| 2022 Março | 53 | 71 | 124 |
| 2022 Fevereiro | 33 | 40 | 73 |
| 2022 Janeiro | 51 | 70 | 121 |
| 2021 Dezembro | 35 | 51 | 86 |
| 2021 Novembro | 37 | 52 | 89 |
| 2021 Outubro | 54 | 66 | 120 |
| 2021 Setembro | 44 | 52 | 96 |
| 2021 Agosto | 42 | 58 | 100 |
| 2021 Julho | 35 | 43 | 78 |
| 2021 Junho | 35 | 39 | 74 |
| 2021 Maio | 54 | 81 | 135 |
| 2021 Abril | 186 | 325 | 511 |
| 2021 Março | 139 | 97 | 236 |
| 2021 Fevereiro | 58 | 33 | 91 |
| 2021 Janeiro | 32 | 15 | 47 |
| 2020 Dezembro | 12 | 8 | 20 |
| 2020 Novembro | 1 | 2 | 3 |
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