Case and Research Letters
Angiosarcoma in Chronic Lymphedema (Stewart-Treves Syndrome)
Angiosarcoma en linfedema crónico (síndrome de Stewart-Treves)
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Pliable, erythematous-violaceous plaques with poorly defined borders are shown. B, Lesion on the forearm during the fourth recurrence 3 years before development of the most recent lesion. This lesion is indurated and brownish, and the borders of the erythematous plaque are poorly defined. C, Lesion on the right thigh during the recurrence 6 months before the most recently developed lesion. The multinodular tumor is superficial and ulcerated and has a fleshy appearance.</p>" ] ] ] "autores" => array:1 [ 0 => array:2 [ "autoresLista" => "A. Agustí-Mejias, F. Messeguer, A. Pérez, V. Alegre de Miquel" "autores" => array:4 [ 0 => array:2 [ "nombre" => "A." "apellidos" => "Agustí-Mejias" ] 1 => array:2 [ "nombre" => "F." "apellidos" => "Messeguer" ] 2 => array:2 [ "nombre" => "A." "apellidos" => "Pérez" ] 3 => array:2 [ "nombre" => "V." 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Sánchez-Medina, A. Acosta, J. Vilar, J. Fernández-Palacios" "autores" => array:4 [ 0 => array:4 [ "nombre" => "M.T." "apellidos" => "Sánchez-Medina" "email" => array:1 [ 0 => "marisolsm_@hotmail.com" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "A." "apellidos" => "Acosta" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "J." "apellidos" => "Vilar" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "J." "apellidos" => "Fernández-Palacios" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] ] "afiliaciones" => array:2 [ 0 => array:3 [ "entidad" => "Servicio de Cirugía Plástica Estética y Reparadora, Hospital de Gran Canaria Dr. Negrín, Las Palmas de Gran Canaria, Spain" "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Servicio de Dermatología Médico-Quirúrgica y Venereología, Hospital de Gran Canaria Dr. Negrín, Las Palmas de Gran Canaria, Spain" "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Angiosarcoma en linfedema crónico (síndrome de Stewart-Treves)" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 1206 "Ancho" => 951 "Tamanyo" => 118968 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Clinical image of the reddish-purple lesions on the anterior surface of the right arm.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Angiosarcoma is a malignant endothelial tumor that can form in any part of the body, including the skin. It is known as hemangiosarcoma or lymphangiosarcoma, depending on whether it derives from the endothelium of the blood vessels or the lymph vessels, respectively.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">It was first described in 1948 by Stewart and Treves<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a> in a series of 6 cases of lymphangiosarcoma after chronic postmastectomy lymphedema; since then, more than 400 cases have been reported of angiosarcoma associated with chronic lymphedema at different sites.</p><p id="par0015" class="elsevierStylePara elsevierViewall">We report the case of a patient with angiosarcoma of the right arm, associated with chronic lymphedema due to non-Hodgkin lymphoma.</p><p id="par0020" class="elsevierStylePara elsevierViewall">The only relevant history reported by the 84-year-old woman was a nodule in the lower right cervical region; results of a fine-needle aspiration biopsy of the nodule were negative for malignancy. Because of the growth of the nodule, a new fine-needle aspiration procedure was performed a year later and cytology results indicated suspected lymphoma; resection and biopsy were performed and a diagnosis of non-Hodgkin large B cell (CD20<span class="elsevierStyleSup">+</span>) lymphoma was established.</p><p id="par0025" class="elsevierStylePara elsevierViewall">Computed tomography (CT) revealed several bilateral enlarged axillary lymph nodes; the largest of these, measuring 2.5-3<span class="elsevierStyleHsp" style=""></span>cm, was on the right side and in contact with the rib. The CT scan also revealed multiple enlarged hilar, mediastinal, para-aortic, and retroesophageal lymph nodes, and a 4.3-cm solid mass in the anterior segment of the right upper lobe of the lung.</p><p id="par0030" class="elsevierStylePara elsevierViewall">An atypical segment resection of the right upper lobe and a mediastinal lymphadenectomy were performed, followed by adjuvant chemotherapy with cyclophosphamide, epirubicin, vincristine, and prednisone (6 cycles). Treatment response was complete, except in the right axilla, where the patient developed a palpable mass measuring 5 to 6<span class="elsevierStyleHsp" style=""></span>cm that was fixed to the deeper layers; the mass was painful and produced a cutaneous reaction. The area was treated with external radiotherapy, with a fraction size of 1.8<span class="elsevierStyleHsp" style=""></span>Gy to achieve a dose of 45<span class="elsevierStyleHsp" style=""></span>Gy; the size of the mass decreased considerably in response to treatment.</p><p id="par0035" class="elsevierStylePara elsevierViewall">Nine years later, the woman was referred to our department with lesions on the anterior surface of the right arm, at the site of the chronic lymphedema; the lesions had appeared 2 weeks earlier and had begun as reddish-purple macules that transformed into plaques and, finally, gave rise to a tumor measuring 16<span class="elsevierStyleHsp" style=""></span>×<span class="elsevierStyleHsp" style=""></span>6<span class="elsevierStyleHsp" style=""></span>cm. She also had erythematous-violaceous lesions measuring 1<span class="elsevierStyleHsp" style=""></span>cm in diameter which had recently appeared on the outer quadrants of the right breast (<a class="elsevierStyleCrossRefs" href="#fig0005">Figs. 1 and 2</a>). Histopathology of the lesion on the arm and those on the breast was compatible with lymphangiosarcoma and showed clefts between the collagen bundles, delineated by spindle-shaped endothelial cells and atypical hyperchromatic epithelial cells. Immunohistochemistry was positive for CD34, CD31, and factor VIII, thereby supporting the histologic diagnosis<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a> (<a class="elsevierStyleCrossRef" href="#fig0015">Fig. 3</a>). The staging study was negative.</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0040" class="elsevierStylePara elsevierViewall">After evaluation by the soft-tissue sarcoma multidisciplinary committee, it was decided to treat the patient with palliative chemotherapy.</p><p id="par0045" class="elsevierStylePara elsevierViewall">Angiosarcoma that develops on a limb with chronic lymphedema or Stewart-Treves syndrome is a rare complication with a rapid and aggressive course.<a class="elsevierStyleCrossRef" href="#bib0020"><span class="elsevierStyleSup">4</span></a> No cure exists to date and the disease is associated with poor prognosis and considerable morbidity. When the disease occurs in the context of a mastectomy, removal of the axillary lymph nodes and radiotherapy are thought to be predisposing factors.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a> The course of the disease is usually aggressive, with rapid local or regional growth of lesions and a tendency toward early distant metastasis.<a class="elsevierStyleCrossRefs" href="#bib0030"><span class="elsevierStyleSup">6,7</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Biopsy is essential for diagnosis and fine-needle aspiration is inadequate. Imaging studies, such as nuclear magnetic resonance imaging, are advisable in order to evaluate local spread.</p><p id="par0055" class="elsevierStylePara elsevierViewall">It is essential to highlight the importance of regular clinical examinations of all patients with chronic lymphedema. While the lesion was first reported in lymphedema secondary to breast cancer, it may occur in any primary or secondary lymphedema. The limbs are the most commonly affected areas, but lymphangiosarcoma has even been reported after abdominoplasty in obese patients.<a class="elsevierStyleCrossRef" href="#bib0040"><span class="elsevierStyleSup">8</span></a> Furthermore, according to published research, the mean time between development of lymphedema and diagnosis of angiosarcoma is between 8 and 10 years; it is thus important to carry out long-term follow-up of patients.<a class="elsevierStyleCrossRefs" href="#bib0020"><span class="elsevierStyleSup">4,6</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">When unusual lesions are found on a limb with lymphedema, histologic studies should be performed to detect this complication early on. Early diagnosis together with radical surgery and associated radiation therapy may increase the chance of survival in these patients. Amputation or wide local excision provides the best chance of long-term survival in patients with Stewart-Treves syndrome. Some ex vivo sensitivity studies have shown that chemotherapy appears to be an adequate complementary approach.<a class="elsevierStyleCrossRefs" href="#bib0045"><span class="elsevierStyleSup">9,10</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">Prevention of lymphedema of the limbs, when treatable secondary causes exist, should be considered essential and existing medical measures to achieve this goal should be optimized.</p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: M.T. Sánchez-Medina, A. Acosta, J. Vilar, J. Fernández-Palacios. Angiosarcoma en linfedema crónico (síndrome de Stewart-Treves). Actas Dermosifiliogr. 2012;103:545-7.</p>" ] ] "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" => 1206 "Ancho" => 951 "Tamanyo" => 118968 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Clinical image of the reddish-purple lesions on the anterior surface of the right arm.</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" => 714 "Ancho" => 951 "Tamanyo" => 69401 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Clinical image of the erythematous-violaceous lesions on the outer quadrants of the right breast.</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" => 976 "Ancho" => 1301 "Tamanyo" => 506434 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0015" class="elsevierStyleSimplePara elsevierViewall">A) Staining with hematoxylin-eosin, original magnification ×40; B) Staining with hematoxylin-eosin, original magnification ×200; C) CD34, original magnification ×100; D) CD31, original magnification ×200.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:10 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "The risk of angiosarcoma following primary breast cancer" "autores" => array:1 [ 0 => array:2 [ "etal" => false "autores" => array:5 [ 0 => "W. 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| año/Mes | Html | Total | |
|---|---|---|---|
| 2024 Noviembre | 8 | 9 | 17 |
| 2024 Octubre | 108 | 53 | 161 |
| 2024 Septiembre | 101 | 45 | 146 |
| 2024 Agosto | 157 | 63 | 220 |
| 2024 Julio | 120 | 55 | 175 |
| 2024 Junio | 122 | 56 | 178 |
| 2024 Mayo | 108 | 49 | 157 |
| 2024 Abril | 98 | 49 | 147 |
| 2024 Marzo | 126 | 49 | 175 |
| 2024 Febrero | 101 | 36 | 137 |
| 2024 Enero | 99 | 44 | 143 |
| 2023 Diciembre | 78 | 29 | 107 |
| 2023 Noviembre | 73 | 42 | 115 |
| 2023 Octubre | 83 | 30 | 113 |
| 2023 Septiembre | 95 | 43 | 138 |
| 2023 Agosto | 86 | 33 | 119 |
| 2023 Julio | 96 | 51 | 147 |
| 2023 Junio | 72 | 29 | 101 |
| 2023 Mayo | 88 | 37 | 125 |
| 2023 Abril | 79 | 38 | 117 |
| 2023 Marzo | 97 | 43 | 140 |
| 2023 Febrero | 77 | 36 | 113 |
| 2023 Enero | 70 | 44 | 114 |
| 2022 Diciembre | 55 | 57 | 112 |
| 2022 Noviembre | 45 | 41 | 86 |
| 2022 Octubre | 44 | 32 | 76 |
| 2022 Septiembre | 42 | 52 | 94 |
| 2022 Agosto | 41 | 57 | 98 |
| 2022 Julio | 29 | 36 | 65 |
| 2022 Junio | 31 | 28 | 59 |
| 2022 Mayo | 60 | 33 | 93 |
| 2022 Abril | 58 | 55 | 113 |
| 2022 Marzo | 71 | 56 | 127 |
| 2022 Febrero | 53 | 45 | 98 |
| 2022 Enero | 45 | 49 | 94 |
| 2021 Diciembre | 69 | 52 | 121 |
| 2021 Noviembre | 67 | 50 | 117 |
| 2021 Octubre | 71 | 65 | 136 |
| 2021 Septiembre | 64 | 41 | 105 |
| 2021 Agosto | 87 | 40 | 127 |
| 2021 Julio | 42 | 35 | 77 |
| 2021 Junio | 70 | 28 | 98 |
| 2021 Mayo | 47 | 46 | 93 |
| 2021 Abril | 80 | 73 | 153 |
| 2021 Marzo | 69 | 30 | 99 |
| 2021 Febrero | 87 | 37 | 124 |
| 2021 Enero | 68 | 22 | 90 |
| 2020 Diciembre | 54 | 31 | 85 |
| 2020 Noviembre | 36 | 23 | 59 |
| 2020 Octubre | 42 | 19 | 61 |
| 2020 Septiembre | 64 | 12 | 76 |
| 2020 Agosto | 41 | 20 | 61 |
| 2020 Julio | 36 | 18 | 54 |
| 2020 Junio | 31 | 22 | 53 |
| 2020 Mayo | 34 | 19 | 53 |
| 2020 Abril | 32 | 23 | 55 |
| 2020 Marzo | 29 | 21 | 50 |
| 2020 Febrero | 6 | 2 | 8 |
| 2020 Enero | 6 | 2 | 8 |
| 2019 Diciembre | 8 | 4 | 12 |
| 2019 Noviembre | 4 | 0 | 4 |
| 2019 Octubre | 0 | 1 | 1 |
| 2019 Septiembre | 4 | 2 | 6 |
| 2019 Agosto | 4 | 1 | 5 |
| 2019 Julio | 4 | 3 | 7 |
| 2019 Junio | 4 | 11 | 15 |
| 2019 Mayo | 7 | 19 | 26 |
| 2019 Abril | 2 | 18 | 20 |
| 2019 Marzo | 4 | 9 | 13 |
| 2019 Febrero | 0 | 4 | 4 |
| 2019 Enero | 4 | 4 | 8 |
| 2018 Diciembre | 2 | 5 | 7 |
| 2018 Noviembre | 2 | 2 | 4 |
| 2018 Octubre | 2 | 0 | 2 |
| 2018 Septiembre | 4 | 0 | 4 |
| 2018 Julio | 0 | 1 | 1 |
| 2018 Junio | 0 | 1 | 1 |
| 2018 Mayo | 0 | 6 | 6 |
| 2018 Abril | 0 | 1 | 1 |
| 2018 Marzo | 1 | 0 | 1 |
| 2018 Febrero | 87 | 11 | 98 |
| 2018 Enero | 96 | 8 | 104 |
| 2017 Diciembre | 93 | 12 | 105 |
| 2017 Noviembre | 82 | 5 | 87 |
| 2017 Octubre | 96 | 2 | 98 |
| 2017 Septiembre | 63 | 18 | 81 |
| 2017 Agosto | 59 | 13 | 72 |
| 2017 Julio | 51 | 8 | 59 |
| 2017 Junio | 60 | 15 | 75 |
| 2017 Mayo | 63 | 10 | 73 |
| 2017 Abril | 59 | 8 | 67 |
| 2017 Marzo | 39 | 35 | 74 |
| 2017 Febrero | 127 | 6 | 133 |
| 2017 Enero | 63 | 15 | 78 |
| 2016 Diciembre | 81 | 8 | 89 |
| 2016 Noviembre | 108 | 13 | 121 |
| 2016 Octubre | 140 | 21 | 161 |
| 2016 Septiembre | 202 | 12 | 214 |
| 2016 Agosto | 110 | 18 | 128 |
| 2016 Julio | 58 | 24 | 82 |
| 2016 Junio | 10 | 16 | 26 |
| 2016 Mayo | 8 | 17 | 25 |
| 2016 Abril | 5 | 8 | 13 |
| 2016 Marzo | 5 | 4 | 9 |
| 2016 Febrero | 9 | 5 | 14 |
| 2016 Enero | 17 | 7 | 24 |
| 2015 Diciembre | 16 | 1 | 17 |
| 2015 Noviembre | 20 | 5 | 25 |
| 2015 Octubre | 32 | 1 | 33 |
| 2015 Septiembre | 32 | 1 | 33 |
| 2015 Agosto | 22 | 5 | 27 |
| 2015 Julio | 169 | 9 | 178 |
| 2015 Junio | 78 | 4 | 82 |
| 2015 Mayo | 113 | 15 | 128 |
| 2015 Abril | 115 | 8 | 123 |
| 2015 Marzo | 123 | 10 | 133 |
| 2015 Febrero | 99 | 5 | 104 |
| 2015 Enero | 87 | 10 | 97 |
| 2014 Diciembre | 78 | 11 | 89 |
| 2014 Noviembre | 77 | 10 | 87 |
| 2014 Octubre | 96 | 15 | 111 |
| 2014 Septiembre | 90 | 10 | 100 |
| 2014 Agosto | 89 | 18 | 107 |
| 2014 Julio | 100 | 21 | 121 |
| 2014 Junio | 65 | 2 | 67 |
| 2014 Mayo | 71 | 8 | 79 |
| 2014 Abril | 56 | 4 | 60 |
| 2014 Marzo | 56 | 10 | 66 |
| 2014 Febrero | 61 | 9 | 70 |
| 2014 Enero | 60 | 10 | 70 |
| 2013 Diciembre | 53 | 9 | 62 |
| 2013 Noviembre | 55 | 8 | 63 |
| 2013 Octubre | 38 | 5 | 43 |
| 2013 Septiembre | 43 | 3 | 46 |
| 2013 Agosto | 28 | 9 | 37 |
| 2013 Julio | 11 | 6 | 17 |
| 2013 Junio | 21 | 22 | 43 |
| 2013 Mayo | 15 | 7 | 22 |
| 2013 Abril | 8 | 17 | 25 |
| 2013 Marzo | 12 | 18 | 30 |
| 2013 Febrero | 37 | 5 | 42 |
| 2013 Enero | 87 | 10 | 97 |
| 2012 Diciembre | 42 | 4 | 46 |
| 2012 Noviembre | 2 | 3 | 5 |
| 2012 Octubre | 2 | 7 | 9 |
| 2012 Septiembre | 0 | 5 | 5 |
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