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only 78 cases were described in the literature&#59; Most patients are white&#44; female &#40;2&#58; 1&#41; and in the second decade of life &#40;average age of 16-17 years old&#41;&#46; Translocation in the EWSR1 gene on chromosome 22q12 has been demonstrated&#44; as well as in situ fluorescence hybridization &#47; EWSR1 with a positive break for the translocation of EWSR1&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2&#44;6</span></a></p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Clinical case</span><p id="par0015" class="elsevierStylePara elsevierViewall">A 25-year old female with 22-days postpartum consulted presenting a 7-month history of a rapidly growing mass on the right thigh&#46; The patient indicates fatigue and associated pain and bleeding of the mass&#46; Physical examination revealed a 5-cm firm erythematous mass&#44; with hematic crust on the surface surrounded with a peau d&#39;orange erythematous halo&#46; &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">A punch biopsy was performed showing a basophilic small-cell tumor&#44; with scarce cytoplasm&#44; prominent nucleus&#44; and chromatin&#44; cells arranged in blankets throughout the thickness of the dermis without epidermal compromise &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Immunohistochemical study was positive for CD99&#44; PKC&#44; and Vimentin&#59; and negative for actin&#44; desmin&#44; S100&#44; chromogranin&#44; CD20&#44; CD3 and CK20&#46; Thoracic CT revealed a 5&#8239;mm solid non-calcic nodule at the base of the left lung&#44; suspected as metastasis&#44; and left pachypleuritis&#46; Abdominal CT showed hepatomegaly at the expense of the right lobe&#44; without dilatation of bile ducts&#44; no solid or cystic masses in the parenchyma where shown&#46; Splenomegaly with a hypodense heterogeneous image&#44; no greater than 25&#8239;mm was found&#44; also suspected as metastasis&#46; A diagnosis of Primitive Neuroectodermal Tumor&#58; Ewing&#39;s Primary Cutaneous Sarcoma was given&#46; The patient was referred to a social-security hospital where she received one induction cycle with vincristine and doxorubicin obtaining 50&#37; reduction of the mass&#46; Following two cycles without vincristine because supplies ran out&#46; Bleeding presented with the third cycle&#44; therefore radiotherapy was indicated &#40;linear accelerator&#41;&#46; Six months later&#44; there is a 90&#37; reduction in the skin lesion &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; Lamentably&#44; the patient died two weeks after the last dermatology appointment&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion</span><p id="par0025" class="elsevierStylePara elsevierViewall">The Ewing&#39;s Sarcoma tumor family conventionally includes three significant entities&#58; Ewing&#39;s sarcoma&#44; Askin&#39;s tumor of the chest wall&#44; and PNET&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;7</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Ewing&#39;s Sarcoma is a primitive neuroectodermal tumor that rarely occurs in the skin and subcutaneous tissue&#46; 85&#37; of tumors present EWSR1 gene fusion with an erythroblastosis virus-transforming gene &#40;FLI1&#41;&#59; the fusion of exon 7 from EWSR1 to exon 6 of FLI1 translocation is specific to Ewing&#39;s Sarcoma&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> In general&#44; it is better known as a primary bone tumor with a higher incidence in children than in adults&#46; It is the second most common primary bone tumor in children and adolescents&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;7</span></a> Ewing&#39;s sarcoma has a low frequency&#44; described mostly in the deep soft tissues of the paraspinal region&#44; the chest wall or the lower extremities&#46; The most superficial cases&#44; called cutaneous&#44; are sporadic and the vast majority of these have been reported as a single&#44; small mass&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Histologically&#44; these sarcomas are composed mainly of small&#44; round tumoral cells that generally express CD99 and weak positivity for synaptophysin markers&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#8211;6&#44;8</span></a> Some small round cell pediatric tumors with skin involvement can be easily ruled out by immunohistochemistry&#46; Rhabdomyosarcoma may show positivity for CD99&#44; but it also stains muscle markers such as desmin&#44; myogenin&#44; and myo-D1&#44; while tumors of the Ewing&#39;s Sarcoma family do not&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#8211;8</span></a> Lymphoblastic lymphoma may have skin involvement and often shows CD99 immunostaining but also Tdt stain&#44; whereas the Tumor of the Ewing&#39;s Sarcoma family is always negative&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;4&#44;6&#44;7</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Primary Cutaneous Ewing Sarcoma is a rare entity&#44; currently treated in the same way as Ewing&#39;s Sarcoma of bone using extensive surgery&#44; radiotherapy&#44; and multi-drug chemotherapy&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;4</span></a> The cutaneous disease presents a slow course and a favorable prognosis&#44; with a survival rate of 91&#37; in ten years&#44; compared with Ewing sarcoma of bone or soft tissue&#44; which have a worse prognosis&#46; The less aggressive behavior of the cutaneous clinical picture probably occurs due to superficial location&#44; smaller tumors&#44; and easy access&#59; allowing detection through routine clinical examination or by the patient providing early diagnosis and complete surgical resection&#44; avoiding the onset of metastasis&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2&#44;4&#44;7</span></a> If the disease is detected early and managed with local excision and systemic chemotherapy&#44; the result is favorable&#46; Nowadays there is no scientific literature that proves a direct and causal relation between Primary Cutaneous Ewing&#39;s Sarcoma and Pregnancy&#46; Unfortunately&#44; as the case we present&#44; 11&#37; of cases of primary cutaneous Ewing sarcoma have described metastatic disease during the presentation&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#8211;8</span></a></p></span></span>"
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Case and Research Letters
Ewing's Primary Cutaneous Sarcoma in a Pregnant Patient: A Case Report
Sarcoma de Ewing Cutáneo Primario en una Paciente Embarazada: informe de un caso caso
Marielos Posada
Autor para correspondencia
marielosposada@ufm.edu

Corresponding author.
, Gabriela Villalta, María-Sofía Roldán, Helga Sarti
Residente en Dermatología, Instituto de Dermatología y Cirugía de Piel (INDERMA), Ciudad de Guatemala, Guatemala
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    "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">The term comes from James Ewing &#40;1866-1943&#41; who was the first to describe the tumor&#44; establishing that the disease was different from lymphoma and described it first as malignant bone endothelioma&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">Angervall and Enzinger identified that the tumor could occur in deep soft tissue&#44; and rarely in the skin&#46;<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> Later&#44; cytogenetic studies confirmed that these tumors belong to the same family as Ewing&#39;s Sarcoma that arises from the bone&#44; commonly called tumors of the Ewing&#39;s Sarcoma family &#40;ESFT&#41;&#46;<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">7</span></a> Until 2001&#44; only 78 cases were described in the literature&#59; Most patients are white&#44; female &#40;2&#58; 1&#41; and in the second decade of life &#40;average age of 16-17 years old&#41;&#46; Translocation in the EWSR1 gene on chromosome 22q12 has been demonstrated&#44; as well as in situ fluorescence hybridization &#47; EWSR1 with a positive break for the translocation of EWSR1&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2&#44;6</span></a></p><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Clinical case</span><p id="par0015" class="elsevierStylePara elsevierViewall">A 25-year old female with 22-days postpartum consulted presenting a 7-month history of a rapidly growing mass on the right thigh&#46; The patient indicates fatigue and associated pain and bleeding of the mass&#46; Physical examination revealed a 5-cm firm erythematous mass&#44; with hematic crust on the surface surrounded with a peau d&#39;orange erythematous halo&#46; &#40;<a class="elsevierStyleCrossRef" href="#fig0005">Fig&#46; 1</a>&#41;&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">A punch biopsy was performed showing a basophilic small-cell tumor&#44; with scarce cytoplasm&#44; prominent nucleus&#44; and chromatin&#44; cells arranged in blankets throughout the thickness of the dermis without epidermal compromise &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Immunohistochemical study was positive for CD99&#44; PKC&#44; and Vimentin&#59; and negative for actin&#44; desmin&#44; S100&#44; chromogranin&#44; CD20&#44; CD3 and CK20&#46; Thoracic CT revealed a 5&#8239;mm solid non-calcic nodule at the base of the left lung&#44; suspected as metastasis&#44; and left pachypleuritis&#46; Abdominal CT showed hepatomegaly at the expense of the right lobe&#44; without dilatation of bile ducts&#44; no solid or cystic masses in the parenchyma where shown&#46; Splenomegaly with a hypodense heterogeneous image&#44; no greater than 25&#8239;mm was found&#44; also suspected as metastasis&#46; A diagnosis of Primitive Neuroectodermal Tumor&#58; Ewing&#39;s Primary Cutaneous Sarcoma was given&#46; The patient was referred to a social-security hospital where she received one induction cycle with vincristine and doxorubicin obtaining 50&#37; reduction of the mass&#46; Following two cycles without vincristine because supplies ran out&#46; Bleeding presented with the third cycle&#44; therefore radiotherapy was indicated &#40;linear accelerator&#41;&#46; Six months later&#44; there is a 90&#37; reduction in the skin lesion &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41;&#46; Lamentably&#44; the patient died two weeks after the last dermatology appointment&#46;</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Discussion</span><p id="par0025" class="elsevierStylePara elsevierViewall">The Ewing&#39;s Sarcoma tumor family conventionally includes three significant entities&#58; Ewing&#39;s sarcoma&#44; Askin&#39;s tumor of the chest wall&#44; and PNET&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;7</span></a></p><p id="par0030" class="elsevierStylePara elsevierViewall">Ewing&#39;s Sarcoma is a primitive neuroectodermal tumor that rarely occurs in the skin and subcutaneous tissue&#46; 85&#37; of tumors present EWSR1 gene fusion with an erythroblastosis virus-transforming gene &#40;FLI1&#41;&#59; the fusion of exon 7 from EWSR1 to exon 6 of FLI1 translocation is specific to Ewing&#39;s Sarcoma&#46;<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> In general&#44; it is better known as a primary bone tumor with a higher incidence in children than in adults&#46; It is the second most common primary bone tumor in children and adolescents&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;7</span></a> Ewing&#39;s sarcoma has a low frequency&#44; described mostly in the deep soft tissues of the paraspinal region&#44; the chest wall or the lower extremities&#46; The most superficial cases&#44; called cutaneous&#44; are sporadic and the vast majority of these have been reported as a single&#44; small mass&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2</span></a></p><p id="par0035" class="elsevierStylePara elsevierViewall">Histologically&#44; these sarcomas are composed mainly of small&#44; round tumoral cells that generally express CD99 and weak positivity for synaptophysin markers&#46;<a class="elsevierStyleCrossRefs" href="#bib0015"><span class="elsevierStyleSup">3&#8211;6&#44;8</span></a> Some small round cell pediatric tumors with skin involvement can be easily ruled out by immunohistochemistry&#46; Rhabdomyosarcoma may show positivity for CD99&#44; but it also stains muscle markers such as desmin&#44; myogenin&#44; and myo-D1&#44; while tumors of the Ewing&#39;s Sarcoma family do not&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#8211;8</span></a> Lymphoblastic lymphoma may have skin involvement and often shows CD99 immunostaining but also Tdt stain&#44; whereas the Tumor of the Ewing&#39;s Sarcoma family is always negative&#46;<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2&#44;4&#44;6&#44;7</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Primary Cutaneous Ewing Sarcoma is a rare entity&#44; currently treated in the same way as Ewing&#39;s Sarcoma of bone using extensive surgery&#44; radiotherapy&#44; and multi-drug chemotherapy&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;4</span></a> The cutaneous disease presents a slow course and a favorable prognosis&#44; with a survival rate of 91&#37; in ten years&#44; compared with Ewing sarcoma of bone or soft tissue&#44; which have a worse prognosis&#46; The less aggressive behavior of the cutaneous clinical picture probably occurs due to superficial location&#44; smaller tumors&#44; and easy access&#59; allowing detection through routine clinical examination or by the patient providing early diagnosis and complete surgical resection&#44; avoiding the onset of metastasis&#46;<a class="elsevierStyleCrossRefs" href="#bib0005"><span class="elsevierStyleSup">1&#44;2&#44;4&#44;7</span></a> If the disease is detected early and managed with local excision and systemic chemotherapy&#44; the result is favorable&#46; Nowadays there is no scientific literature that proves a direct and causal relation between Primary Cutaneous Ewing&#39;s Sarcoma and Pregnancy&#46; Unfortunately&#44; as the case we present&#44; 11&#37; of cases of primary cutaneous Ewing sarcoma have described metastatic disease during the presentation&#46;<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6&#8211;8</span></a></p></span></span>"
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