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
Corresponding author
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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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 17 | 9 | 26 |
| 2024 October | 89 | 73 | 162 |
| 2024 September | 136 | 70 | 206 |
| 2024 August | 150 | 105 | 255 |
| 2024 July | 147 | 100 | 247 |
| 2024 June | 96 | 88 | 184 |
| 2024 May | 83 | 62 | 145 |
| 2024 April | 94 | 89 | 183 |
| 2024 March | 76 | 66 | 142 |
| 2024 February | 80 | 76 | 156 |
| 2024 January | 59 | 47 | 106 |
| 2023 December | 53 | 55 | 108 |
| 2023 November | 61 | 77 | 138 |
| 2023 October | 50 | 71 | 121 |
| 2023 September | 67 | 63 | 130 |
| 2023 August | 66 | 63 | 129 |
| 2023 July | 45 | 115 | 160 |
| 2023 June | 53 | 38 | 91 |
| 2023 May | 47 | 26 | 73 |
| 2023 April | 33 | 36 | 69 |
| 2023 March | 53 | 28 | 81 |
| 2023 February | 37 | 34 | 71 |
| 2023 January | 46 | 61 | 107 |
| 2022 December | 51 | 42 | 93 |
| 2022 November | 51 | 56 | 107 |
| 2022 October | 66 | 65 | 131 |
| 2022 September | 52 | 115 | 167 |
| 2022 August | 35 | 69 | 104 |
| 2022 July | 40 | 56 | 96 |
| 2022 June | 40 | 86 | 126 |
| 2022 May | 36 | 55 | 91 |
| 2022 April | 38 | 53 | 91 |
| 2022 March | 53 | 71 | 124 |
| 2022 February | 33 | 40 | 73 |
| 2022 January | 51 | 70 | 121 |
| 2021 December | 35 | 51 | 86 |
| 2021 November | 37 | 52 | 89 |
| 2021 October | 54 | 66 | 120 |
| 2021 September | 44 | 52 | 96 |
| 2021 August | 42 | 58 | 100 |
| 2021 July | 35 | 43 | 78 |
| 2021 June | 35 | 39 | 74 |
| 2021 May | 54 | 81 | 135 |
| 2021 April | 186 | 325 | 511 |
| 2021 March | 139 | 97 | 236 |
| 2021 February | 58 | 33 | 91 |
| 2021 January | 32 | 15 | 47 |
| 2020 December | 12 | 8 | 20 |
| 2020 November | 1 | 2 | 3 |
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