Case for Diagnosis
Skin Tumors Arising on Chronic Lymphedema
Lesiones tumorales cutáneas sobre linfedema crónico
I. Latour-Álvarez
, C. Vázquez-Rodríguez, M. García-Bustínduy
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Departamento de Dermatología, Hospital Universitario de Canarias, Santa Cruz de Tenerife, Islas Canarias, España
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"apellidos" => "García-Bustínduy" ] ] "afiliaciones" => array:1 [ 0 => array:2 [ "entidad" => "Departamento de Dermatología, Hospital Universitario de Canarias, Santa Cruz de Tenerife, Islas Canarias, España" "identificador" => "aff0005" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Lesiones tumorales cutáneas sobre linfedema crónico" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:6 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 458 "Ancho" => 850 "Tamanyo" => 73508 ] ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><span id="sec0005" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0005">Medical History</span><p id="par0005" class="elsevierStylePara elsevierViewall">An 88-year-old woman with dependence in activities of daily living presented with chronic lymphedema secondary to left breast cancer treated with radical mastectomy, ipsilateral lymphadenectomy, radiotherapy, and chemotherapy 10 years earlier. Four months before this visit, she started to develop hematoma-like lesions on her left forearm. The lesions grew fast, causing pain and bleeding and prompting her to visit the emergency department, where she was evaluated by a dermatologist.</p></span><span id="sec0010" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0010">Physical Examination</span><p id="par0010" class="elsevierStylePara elsevierViewall">Edematous right upper limb with multiple edematous, violaceous tumor lesions with a diameter of 3<span class="elsevierStyleHsp" style=""></span>cm to 10<span class="elsevierStyleHsp" style=""></span>cm. The lesions had an elastic-like consistency and some of them were ulcerated and bleeding (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>). In the left chest area, near the mastectomy scar, there were 2 isolated 1-cm lesions with similar characteristics.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia></span><span id="sec0015" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0015">Histopathology</span><p id="par0015" class="elsevierStylePara elsevierViewall">The histopathologic study showed a dermal sheetlike proliferation of pleomorphic cells, many of which formed a solid pattern. The cells were round and had vesicular nuclei and prominent eosinophilic nucleoli. Abundant mitoses and hematic extravasation were also observed (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>). Immunohistochemical staining was positive for CD34 (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>) and ERG, and negative for cytokeratin, protein S100, GATA3, myeloperoxidase, CD56, and human herpesvirus 8. A genetic alteration in the <span class="elsevierStyleItalic">c-Myc</span> oncogene (chromosome 8q24.21) was detected by fluorescent in situ hybridization.</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0020" class="elsevierStylePara elsevierViewall">What is your diagnosis?</p></span><span id="sec0020" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0020">Diagnosis</span><p id="par0025" class="elsevierStylePara elsevierViewall">Lymphangiosarcoma associated with chronic lymphedema (Stewart-Treves syndrome)</p></span><span id="sec0025" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0025">Clinical Course and Treatment</span><p id="par0030" class="elsevierStylePara elsevierViewall">Following consultation with the Sarcoma Committee, the option of intensive treatment was rejected due to the patient's baseline characteristics. The patient died 1 month after diagnosis.</p></span><span id="sec0030" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0030">Comment</span><p id="par0035" class="elsevierStylePara elsevierViewall">Angiosarcoma is an uncommon tumor derived from the endothelium of lymph and blood vessels. In 1948, Stewart and Treves<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">1</span></a> described an association between angiosarcoma and chronic lymphedema following lymphadenectomy due to breast cancer. Stewart-Treves syndrome has an estimated incidence of between 0.07% and 0.45%. As in our case, it tends to present with multifocal, normally ipsilateral, lesions several years after lymphadenectomy for breast cancer. The mean time to onset is approximately 10 years and the mean age of presentation is 60 years.<a class="elsevierStyleCrossRef" href="#bib0030"><span class="elsevierStyleSup">2</span></a> Although the etiology and pathogenesis are not well established, current pathogenic theories hold that lymphedema, regardless of its cause (radiation, surgery, or other), triggers an angiogenic stimulus in the area of the edema leading to the formation of new collateral lymph and blood vessels. The presence of this continuous angiogenic stimulus might then trigger the appearance of malignancies, such as Stewart-Treves syndrome or Kaposi sarcoma.<a class="elsevierStyleCrossRef" href="#bib0035"><span class="elsevierStyleSup">3</span></a></p><p id="par0040" class="elsevierStylePara elsevierViewall">Angiosarcoma lesions associated with chronic lymphedema or secondary to radiotherapy have relatively consistent genetic alterations. The most common alterations are amplifications in chromosome 8q24.21 (50%), 10p12.33 (33%), and 5q35.3 (11%).<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">4</span></a></p><p id="par0045" class="elsevierStylePara elsevierViewall">The main entities that should be contemplated in the differential diagnosis are Kaposi sarcoma, metastases from breast cancer, and melanoma. Treatment options are limited. Radical surgery, chemotherapy, and immunotherapy have all been used, but they do not appear to have any impact on survival. Prognosis is very poor and mean survival is 20 months after diagnosis.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">2,3</span></a> We have presented a new case of Stewart-Treves syndrome, which, while uncommon, is important to keep in mind considering the high number of lymphadenectomies performed.</p></span><span id="sec0035" class="elsevierStyleSection elsevierViewall"><span class="elsevierStyleSectionTitle" id="sect0035">Conflicts of Interest</span><p id="par0050" class="elsevierStylePara elsevierViewall">The authors declare that they have no conflicts of interest.</p></span></span>" "textoCompletoSecciones" => array:1 [ "secciones" => array:8 [ 0 => array:2 [ "identificador" => "sec0005" "titulo" => "Medical History" ] 1 => array:2 [ "identificador" => "sec0010" "titulo" => "Physical Examination" ] 2 => array:2 [ "identificador" => "sec0015" "titulo" => "Histopathology" ] 3 => array:2 [ "identificador" => "sec0020" "titulo" => "Diagnosis" ] 4 => array:2 [ "identificador" => "sec0025" "titulo" => "Clinical Course and Treatment" ] 5 => array:2 [ "identificador" => "sec0030" "titulo" => "Comment" ] 6 => array:2 [ "identificador" => "sec0035" "titulo" => "Conflicts of Interest" ] 7 => array:1 [ "titulo" => "Références" ] ] ] "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara" id="npar0005">Please cite this article as: Latour-Álvarez I, Vázquez-Rodríguez C, García-Bustínduy M. Lesiones tumorales cutáneas sobre linfedema crónico. Actas Dermosifiliogr. 2018;109:439–440.</p>" ] ] "multimedia" => array:3 [ 0 => array:6 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 458 "Ancho" => 850 "Tamanyo" => 73508 ] ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 485 "Ancho" => 850 "Tamanyo" => 181550 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Hematoxylin-eosin, original magnification ×40.</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" => 632 "Ancho" => 850 "Tamanyo" => 221116 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">CD34 immunohistochemical staining (original magnification ×10).</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "Références" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0015" "bibliografiaReferencia" => array:4 [ 0 => array:3 [ "identificador" => "bib0025" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Lymphangiosarcoma in postmastectomy lymphedema: A report of six cases in elephantiasis chirurgica" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:2 [ 0 => "F.W. Stewart" 1 => "N. Treves" ] ] ] ] ] "host" => array:1 [ 0 => array:1 [ "Revista" => array:6 [ "tituloSerie" => "Cancer" "fecha" => "1948" "volumen" => "1" "paginaInicial" => "64" "paginaFinal" => "81" "link" => array:1 [ 0 => array:2 [ "url" => "https://www.ncbi.nlm.nih.gov/pubmed/18867440" "web" => "Medline" ] ] ] ] ] ] ] ] 1 => array:3 [ "identificador" => "bib0030" "etiqueta" => "2" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "Stewart-Treves syndrome: A case report and review of the literature" "autores" => array:1 [ 0 => array:2 [ "etal" => true "autores" => array:6 [ 0 => "R. Berebichez-Fridman" 1 => "Y.E. Deutsch" 2 => "T.M. Joyal" 3 => "P.M. Olvera" 4 => "P.W. Benedetto" 5 => "A.E. 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| 2024 Octubre | 79 | 34 | 113 |
| 2024 Septiembre | 67 | 22 | 89 |
| 2024 Agosto | 110 | 47 | 157 |
| 2024 Julio | 81 | 19 | 100 |
| 2024 Junio | 100 | 57 | 157 |
| 2024 Mayo | 86 | 36 | 122 |
| 2024 Abril | 104 | 25 | 129 |
| 2024 Marzo | 117 | 24 | 141 |
| 2024 Febrero | 95 | 42 | 137 |
| 2024 Enero | 83 | 25 | 108 |
| 2023 Diciembre | 111 | 18 | 129 |
| 2023 Noviembre | 143 | 21 | 164 |
| 2023 Octubre | 95 | 20 | 115 |
| 2023 Septiembre | 74 | 32 | 106 |
| 2023 Agosto | 46 | 16 | 62 |
| 2023 Julio | 62 | 32 | 94 |
| 2023 Junio | 60 | 17 | 77 |
| 2023 Mayo | 54 | 29 | 83 |
| 2023 Abril | 70 | 21 | 91 |
| 2023 Marzo | 77 | 24 | 101 |
| 2023 Febrero | 58 | 19 | 77 |
| 2023 Enero | 44 | 27 | 71 |
| 2022 Diciembre | 74 | 46 | 120 |
| 2022 Noviembre | 21 | 24 | 45 |
| 2022 Octubre | 19 | 17 | 36 |
| 2022 Septiembre | 28 | 40 | 68 |
| 2022 Agosto | 35 | 35 | 70 |
| 2022 Julio | 20 | 36 | 56 |
| 2022 Junio | 27 | 26 | 53 |
| 2022 Mayo | 31 | 40 | 71 |
| 2022 Abril | 52 | 41 | 93 |
| 2022 Marzo | 48 | 44 | 92 |
| 2022 Febrero | 48 | 26 | 74 |
| 2022 Enero | 40 | 33 | 73 |
| 2021 Diciembre | 28 | 38 | 66 |
| 2021 Noviembre | 41 | 44 | 85 |
| 2021 Octubre | 36 | 52 | 88 |
| 2021 Septiembre | 31 | 42 | 73 |
| 2021 Agosto | 28 | 29 | 57 |
| 2021 Julio | 24 | 27 | 51 |
| 2021 Junio | 25 | 22 | 47 |
| 2021 Mayo | 43 | 45 | 88 |
| 2021 Abril | 71 | 67 | 138 |
| 2021 Marzo | 87 | 21 | 108 |
| 2021 Febrero | 53 | 35 | 88 |
| 2021 Enero | 25 | 15 | 40 |
| 2020 Diciembre | 48 | 22 | 70 |
| 2020 Noviembre | 38 | 22 | 60 |
| 2020 Octubre | 25 | 11 | 36 |
| 2020 Septiembre | 36 | 14 | 50 |
| 2020 Agosto | 33 | 23 | 56 |
| 2020 Julio | 32 | 16 | 48 |
| 2020 Junio | 42 | 34 | 76 |
| 2020 Mayo | 20 | 24 | 44 |
| 2020 Abril | 33 | 15 | 48 |
| 2020 Marzo | 32 | 19 | 51 |
| 2020 Enero | 4 | 0 | 4 |
| 2019 Diciembre | 4 | 0 | 4 |
| 2019 Noviembre | 4 | 0 | 4 |
| 2019 Septiembre | 4 | 0 | 4 |
| 2019 Agosto | 4 | 0 | 4 |
| 2019 Julio | 4 | 0 | 4 |
| 2019 Junio | 3 | 0 | 3 |
| 2019 Mayo | 4 | 0 | 4 |
| 2019 Abril | 2 | 0 | 2 |
| 2019 Marzo | 2 | 0 | 2 |
| 2018 Diciembre | 4 | 0 | 4 |
| 2018 Noviembre | 5 | 0 | 5 |
| 2018 Octubre | 1 | 0 | 1 |
| 2018 Septiembre | 2 | 0 | 2 |
| 2018 Julio | 1 | 0 | 1 |
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