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ascites&#44; or other cutaneous lesions were found&#46; Histology revealed neoplastic endothelial cells forming channels that dissected collagen fibers&#46; The cells exhibited atypia with loss of nuclear-cytoplasmic ratio&#44; pleomorphism&#44; and abundant mitoses &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Immunohistochemistry was positive for CD31 &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41; and CD34 markers in tumor cells and negative for human herpes virus 8&#46; A diagnosis of angiosarcoma was established&#46; A computed tomography scan of the abdomen revealed dermal tumors infiltrating the subcutaneous tissue as far as the abdominal wall below the umbilicus and mild ascites possibly related to peritoneal carcinomatosis&#46; The patient was referred to the oncology department&#44; where she declined the recommended chemotherapy&#46; One month after diagnosis&#44; she was admitted&#46; Her general condition had declined&#44; she had edema in the lower extremities and evident ascites&#46; Paracentesis confirmed peritoneal infiltration of ascitic fluid secondary to the angiosarcoma&#46; She died 2 weeks later from kidney failure&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Angiosarcoma&#44; or malignant angioendothelioma&#44; is an uncommon tumor of endothelial cells that accounts for only 2&#37; of all sarcomas&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> It is an aggressive tumor with a 5-year survival rate of 50&#37; to 60&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Mean survival after diagnosis ranges from 18 to 28 months&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Three types of angiosarcoma are traditionally described&#46; The first is idiopathic cutaneous angiosarcoma&#44; which appears on the head and scalp of older patients&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> The second and third are angiosarcoma secondary to chronic lymphedema &#40;Stewart-Treves syndrome&#41; and a type that appears in skin exposed to radiotherapy&#46; This tumor generally presents as a lesion that resembles bruising&#44; with erythematous-violaceous nodules&#46; The various types of angiosarcoma have similar histologic features&#46; They are poorly defined tumors that trace irregular paths across large networks of dilated vascular channels&#44; infiltrating dermal and subcutaneous layers&#46; Tumor cells have large&#44; hyperchromatic&#44; pleomorphic nuclei and show a high degree of mitotic activity&#46; CD31 positivity is the most sensitive immunohistochemical finding for establishing a diagnosis&#46; Factor VIII and CD34 are secondary markers&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> The pathogenesis of these tumors is not clearly defined&#46; Some have suggested that elevated serum concentrations of vascular endothelial growth factor or angiopoietin 2&#44; or a decrease in cadherins in the vascular endothelium&#44; may play a role&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> c-Myc amplification has also been detected in angiosarcomas secondary to radiotherapy or lymphedema&#46;<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">6&#44;7</span></a> The role of drug-induced immunosuppression in the development of these tumors is unclear&#46; Treatment requires surgical excision and radiotherapy&#46; No evidence yet suggests that chemotherapy improves the prognosis&#44; but chemotherapy with anthracyclines and taxanes would be the first line of treatment when the disease has metastasized&#46; Twenty cases of angiosarcoma have been described to date in kidney transplant recipients on immunosuppressants&#46; The tumor usually appears in an arteriovenous fistula&#46; Only 5 other cutaneous angiosarcomas have been reported&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3&#44;8&#8211;10</span></a> The case we describe is highly unusual&#44; similar only to one reported in 2011&#44; in which an abdominal angiosarcoma appeared 8 years after a kidney transplant in an immunosuppressed patient&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> Our patient&#39;s tumor did not follow the traditional etiological pattern for this type of tumor&#46; Although she had had a mastectomy&#44; she had never undergone radiotherapy&#46; Moreover&#44; lymphedema was absent from the tumor site&#46; The etiology of angiosarcomas in kidney transplanted patients on long-standing immunosuppressant therapy remains unclear&#44; given the few known cases described&#46; Impaired immune vigilance seems to be implicated&#44; but further study is needed to establish the precise relationship between immunosuppression&#44; 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Case and Research Letters
Abdominal Wall Angiosarcoma in a Patient With Drug-Induced Immunosuppression Following Renal Transplantation
Angiosarcoma de pared abdominal en paciente con inmunosupresión farmacológica por trasplante renal
P. Martína,
Autor para correspondencia
Pablo_ronda@hotmail.com

Corresponding author.
, A. Pulpilloa, R. Cabrerab, J. Conejo-Mira
a Unidad de Gestión Clínica de Dermatología, Hospital Universitario Virgen del Rocío, Sevilla, Spain
b Unidad de Gestión Clínica de Anatomía Patológica, Hospital Universitario Virgen del Rocío, Sevilla, Spain
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ascites&#44; or other cutaneous lesions were found&#46; Histology revealed neoplastic endothelial cells forming channels that dissected collagen fibers&#46; The cells exhibited atypia with loss of nuclear-cytoplasmic ratio&#44; pleomorphism&#44; and abundant mitoses &#40;<a class="elsevierStyleCrossRef" href="#fig0010">Fig&#46; 2</a>&#41;&#46; Immunohistochemistry was positive for CD31 &#40;<a class="elsevierStyleCrossRef" href="#fig0015">Fig&#46; 3</a>&#41; and CD34 markers in tumor cells and negative for human herpes virus 8&#46; A diagnosis of angiosarcoma was established&#46; A computed tomography scan of the abdomen revealed dermal tumors infiltrating the subcutaneous tissue as far as the abdominal wall below the umbilicus and mild ascites possibly related to peritoneal carcinomatosis&#46; The patient was referred to the oncology department&#44; where she declined the recommended chemotherapy&#46; One month after diagnosis&#44; she was admitted&#46; Her general condition had declined&#44; she had edema in the lower extremities and evident ascites&#46; Paracentesis confirmed peritoneal infiltration of ascitic fluid secondary to the angiosarcoma&#46; She died 2 weeks later from kidney failure&#46;</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><elsevierMultimedia ident="fig0010"></elsevierMultimedia><elsevierMultimedia ident="fig0015"></elsevierMultimedia><p id="par0010" class="elsevierStylePara elsevierViewall">Angiosarcoma&#44; or malignant angioendothelioma&#44; is an uncommon tumor of endothelial cells that accounts for only 2&#37; of all sarcomas&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> It is an aggressive tumor with a 5-year survival rate of 50&#37; to 60&#37;&#46;<a class="elsevierStyleCrossRef" href="#bib0055"><span class="elsevierStyleSup">1</span></a> Mean survival after diagnosis ranges from 18 to 28 months&#46;<a class="elsevierStyleCrossRef" href="#bib0060"><span class="elsevierStyleSup">2</span></a> Three types of angiosarcoma are traditionally described&#46; The first is idiopathic cutaneous angiosarcoma&#44; which appears on the head and scalp of older patients&#46;<a class="elsevierStyleCrossRef" href="#bib0065"><span class="elsevierStyleSup">3</span></a> The second and third are angiosarcoma secondary to chronic lymphedema &#40;Stewart-Treves syndrome&#41; and a type that appears in skin exposed to radiotherapy&#46; This tumor generally presents as a lesion that resembles bruising&#44; with erythematous-violaceous nodules&#46; The various types of angiosarcoma have similar histologic features&#46; They are poorly defined tumors that trace irregular paths across large networks of dilated vascular channels&#44; infiltrating dermal and subcutaneous layers&#46; Tumor cells have large&#44; hyperchromatic&#44; pleomorphic nuclei and show a high degree of mitotic activity&#46; CD31 positivity is the most sensitive immunohistochemical finding for establishing a diagnosis&#46; Factor VIII and CD34 are secondary markers&#46;<a class="elsevierStyleCrossRef" href="#bib0070"><span class="elsevierStyleSup">4</span></a> The pathogenesis of these tumors is not clearly defined&#46; Some have suggested that elevated serum concentrations of vascular endothelial growth factor or angiopoietin 2&#44; or a decrease in cadherins in the vascular endothelium&#44; may play a role&#46;<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">5</span></a> c-Myc amplification has also been detected in angiosarcomas secondary to radiotherapy or lymphedema&#46;<a class="elsevierStyleCrossRefs" href="#bib0080"><span class="elsevierStyleSup">6&#44;7</span></a> The role of drug-induced immunosuppression in the development of these tumors is unclear&#46; Treatment requires surgical excision and radiotherapy&#46; No evidence yet suggests that chemotherapy improves the prognosis&#44; but chemotherapy with anthracyclines and taxanes would be the first line of treatment when the disease has metastasized&#46; Twenty cases of angiosarcoma have been described to date in kidney transplant recipients on immunosuppressants&#46; The tumor usually appears in an arteriovenous fistula&#46; Only 5 other cutaneous angiosarcomas have been reported&#46;<a class="elsevierStyleCrossRefs" href="#bib0065"><span class="elsevierStyleSup">3&#44;8&#8211;10</span></a> The case we describe is highly unusual&#44; similar only to one reported in 2011&#44; in which an abdominal angiosarcoma appeared 8 years after a kidney transplant in an immunosuppressed patient&#46;<a class="elsevierStyleCrossRef" href="#bib0095"><span class="elsevierStyleSup">9</span></a> Our patient&#39;s tumor did not follow the traditional etiological pattern for this type of tumor&#46; Although she had had a mastectomy&#44; she had never undergone radiotherapy&#46; Moreover&#44; lymphedema was absent from the tumor site&#46; The etiology of angiosarcomas in kidney transplanted patients on long-standing immunosuppressant therapy remains unclear&#44; given the few known cases described&#46; Impaired immune vigilance seems to be implicated&#44; but further study is needed to establish the precise relationship between immunosuppression&#44; 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ISSN: 15782190
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