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• W2012160521 abstract "A 74-year-old previously healthy woman presented with several months of progressive epigastric discomfort, nausea, anorexia, abdominal distention, and lower-extremity swelling. Laboratory evaluation revealed anemia with low iron and elevated ferritin and thrombocytopenia. Computed tomography (CT) of the abdomen showed innumerable low-attenuation hepatic and splenic lesions with partial occlusion of the portal vein. Additional studies, including CT of the brain and chest, colonoscopy, esophagogastroduodenoscopy, lower-extremity ultrasound, and mammogram, were not revealing. Initial percutaneous core liver biopsy showed granulation tissue without evidence of malignancy.On transfer to a tertiary center, the patient was stable with normal vital signs. Physical examination showed mild jaundice, distended jugular veins, abdominal distention with shifting dullness and hepatomegaly, upper abdominal tenderness, and bilateral lower leg edema. Laboratory values showed a hemoglobin level of 8.8 g/dL (reference range, 12.0-15.5 g/dL), platelet count of 76 × 109 (reference range, 150-450 × 109), a normal basic metabolic panel, alkaline phosphatase level of 290 U/L (reference range, 24-110 U/L), aspartate aminotransferase level of 227 U/L (reference range, 15-41 U/L), alanine aminotransferase level of 127 U/L (reference range, 14-54 U/L), and total bilirubin level of 4.6 mg/dL (reference range, 0.4-1.5 g/dL). International normalized ratio was elevated at 1.5 (reference range, 0.8-1.1), fibrinogen was low at 136 mg/dL (reference range, 213-435 mg/dL), and D-dimer was markedly elevated at 26,693 ng/mL (reference range, <500 ng/mL). Tumor markers were sent and showed an elevated cancer antigen 125 at 94.8 U/mL (reference range, 0-35 U/mL) and normal alpha fetoprotein. Liver ultrasound with Doppler showed normal portal flow. Ultrasound-guided percutaneous core liver biopsies were again nondiagnostic, so laparoscopic-guided wedge biopsy of the hepatic lesions was performed. Pathology showed an infiltrating, vasoformative, and epithelioid to spindled cell tumor with moderate atypia and increased mitotic activity (Figure 1A). The tumor cells demonstrated strong expression of CD31 (Figure 1B) and CD34 antigens, and were negative for pancytokeratin and human herpes virus 8. The Ki67 labeling index was approximately 50%. The histology and immunohistochemical profile was consistent with a high-grade angiosarcoma. Consequently, the patient's clinical condition declined with worsening respiratory status, renal function, liver function, and volume overload. Diuresis was attempted without significant improvement. The patient elected not to undergo further treatment and died shortly thereafter at inpatient hospice.DiscussionMetastatic angiosarcoma, most likely splenic in origin in this case, is a rare and often deadly cancer.1Neuhauser T.S. Derringer G.A. Thompson L.D. et al.Splenic angiosarcoma: a clinicopathologic and immunophenotypic study of 28 cases.Mod Pathol. 2000; 13: 978-987Crossref PubMed Scopus (128) Google Scholar, 2Falk S. Krishnan J. Meis J.M. Primary angiosarcoma of the spleen. A clinicopathologic study of 40 cases.Am J Surg Pathol. 1993; 17: 959-970Crossref PubMed Scopus (186) Google Scholar The low incidence, vague clinical presentation, and heterogeneous macroscopic and microscopic appearance can make the diagnosis challenging. Furthermore, the disease carries a high hemorrhagic risk especially in the setting of thrombocytopenia, making repeated biopsies risky.ConclusionsRadiographic evidence of a complex mass or splenic masses with evidence of metastatic disease, often to the liver, is highly suggestive of splenic angiosarcoma. Typical pathologic findings include epithelioid or spindle cells with cytologic atypia and mitotic activity. They often demonstrate evidence of primitive vascular lumen formation and expression of vascular markers, such as ERG, CD31, CD34, and FVIII.3Kasper D. Braunwald E. Fauci A. Hauser S. Longo D. Jameson J. Harrison's Manual of Medicine.in: 16th McGraw Hill Professional, New York, NY2005Google Scholar Primary splenic angiosarcoma is a rare diagnosis with only 200 cases reported worldwide,4Valbuena J.R. Levenback C. Mansfield P. Liu J. Angiosarcoma of the spleen clinically presenting as metastatic ovarian cancer. A case report and review of the literature.Ann Diagn Pathol. 2005; 9: 289-292Abstract Full Text Full Text PDF PubMed Scopus (22) Google Scholar but it remains the most common nonhematologic malignancy of the spleen. It is highly aggressive with frequent early metastasis, most commonly to the liver. Neither treatment with chemotherapy nor radiation therapy has been shown to improve outcomes; however, splenectomy can improve survival in localized disease. A 74-year-old previously healthy woman presented with several months of progressive epigastric discomfort, nausea, anorexia, abdominal distention, and lower-extremity swelling. Laboratory evaluation revealed anemia with low iron and elevated ferritin and thrombocytopenia. Computed tomography (CT) of the abdomen showed innumerable low-attenuation hepatic and splenic lesions with partial occlusion of the portal vein. Additional studies, including CT of the brain and chest, colonoscopy, esophagogastroduodenoscopy, lower-extremity ultrasound, and mammogram, were not revealing. Initial percutaneous core liver biopsy showed granulation tissue without evidence of malignancy. On transfer to a tertiary center, the patient was stable with normal vital signs. Physical examination showed mild jaundice, distended jugular veins, abdominal distention with shifting dullness and hepatomegaly, upper abdominal tenderness, and bilateral lower leg edema. Laboratory values showed a hemoglobin level of 8.8 g/dL (reference range, 12.0-15.5 g/dL), platelet count of 76 × 109 (reference range, 150-450 × 109), a normal basic metabolic panel, alkaline phosphatase level of 290 U/L (reference range, 24-110 U/L), aspartate aminotransferase level of 227 U/L (reference range, 15-41 U/L), alanine aminotransferase level of 127 U/L (reference range, 14-54 U/L), and total bilirubin level of 4.6 mg/dL (reference range, 0.4-1.5 g/dL). International normalized ratio was elevated at 1.5 (reference range, 0.8-1.1), fibrinogen was low at 136 mg/dL (reference range, 213-435 mg/dL), and D-dimer was markedly elevated at 26,693 ng/mL (reference range, <500 ng/mL). Tumor markers were sent and showed an elevated cancer antigen 125 at 94.8 U/mL (reference range, 0-35 U/mL) and normal alpha fetoprotein. Liver ultrasound with Doppler showed normal portal flow. Ultrasound-guided percutaneous core liver biopsies were again nondiagnostic, so laparoscopic-guided wedge biopsy of the hepatic lesions was performed. Pathology showed an infiltrating, vasoformative, and epithelioid to spindled cell tumor with moderate atypia and increased mitotic activity (Figure 1A). The tumor cells demonstrated strong expression of CD31 (Figure 1B) and CD34 antigens, and were negative for pancytokeratin and human herpes virus 8. The Ki67 labeling index was approximately 50%. The histology and immunohistochemical profile was consistent with a high-grade angiosarcoma. Consequently, the patient's clinical condition declined with worsening respiratory status, renal function, liver function, and volume overload. Diuresis was attempted without significant improvement. The patient elected not to undergo further treatment and died shortly thereafter at inpatient hospice. DiscussionMetastatic angiosarcoma, most likely splenic in origin in this case, is a rare and often deadly cancer.1Neuhauser T.S. Derringer G.A. Thompson L.D. et al.Splenic angiosarcoma: a clinicopathologic and immunophenotypic study of 28 cases.Mod Pathol. 2000; 13: 978-987Crossref PubMed Scopus (128) Google Scholar, 2Falk S. Krishnan J. Meis J.M. Primary angiosarcoma of the spleen. A clinicopathologic study of 40 cases.Am J Surg Pathol. 1993; 17: 959-970Crossref PubMed Scopus (186) Google Scholar The low incidence, vague clinical presentation, and heterogeneous macroscopic and microscopic appearance can make the diagnosis challenging. Furthermore, the disease carries a high hemorrhagic risk especially in the setting of thrombocytopenia, making repeated biopsies risky. Metastatic angiosarcoma, most likely splenic in origin in this case, is a rare and often deadly cancer.1Neuhauser T.S. Derringer G.A. Thompson L.D. et al.Splenic angiosarcoma: a clinicopathologic and immunophenotypic study of 28 cases.Mod Pathol. 2000; 13: 978-987Crossref PubMed Scopus (128) Google Scholar, 2Falk S. Krishnan J. Meis J.M. Primary angiosarcoma of the spleen. A clinicopathologic study of 40 cases.Am J Surg Pathol. 1993; 17: 959-970Crossref PubMed Scopus (186) Google Scholar The low incidence, vague clinical presentation, and heterogeneous macroscopic and microscopic appearance can make the diagnosis challenging. Furthermore, the disease carries a high hemorrhagic risk especially in the setting of thrombocytopenia, making repeated biopsies risky. ConclusionsRadiographic evidence of a complex mass or splenic masses with evidence of metastatic disease, often to the liver, is highly suggestive of splenic angiosarcoma. Typical pathologic findings include epithelioid or spindle cells with cytologic atypia and mitotic activity. They often demonstrate evidence of primitive vascular lumen formation and expression of vascular markers, such as ERG, CD31, CD34, and FVIII.3Kasper D. Braunwald E. Fauci A. Hauser S. Longo D. Jameson J. Harrison's Manual of Medicine.in: 16th McGraw Hill Professional, New York, NY2005Google Scholar Primary splenic angiosarcoma is a rare diagnosis with only 200 cases reported worldwide,4Valbuena J.R. Levenback C. Mansfield P. Liu J. Angiosarcoma of the spleen clinically presenting as metastatic ovarian cancer. A case report and review of the literature.Ann Diagn Pathol. 2005; 9: 289-292Abstract Full Text Full Text PDF PubMed Scopus (22) Google Scholar but it remains the most common nonhematologic malignancy of the spleen. It is highly aggressive with frequent early metastasis, most commonly to the liver. Neither treatment with chemotherapy nor radiation therapy has been shown to improve outcomes; however, splenectomy can improve survival in localized disease. Radiographic evidence of a complex mass or splenic masses with evidence of metastatic disease, often to the liver, is highly suggestive of splenic angiosarcoma. Typical pathologic findings include epithelioid or spindle cells with cytologic atypia and mitotic activity. They often demonstrate evidence of primitive vascular lumen formation and expression of vascular markers, such as ERG, CD31, CD34, and FVIII.3Kasper D. Braunwald E. Fauci A. Hauser S. Longo D. Jameson J. Harrison's Manual of Medicine.in: 16th McGraw Hill Professional, New York, NY2005Google Scholar Primary splenic angiosarcoma is a rare diagnosis with only 200 cases reported worldwide,4Valbuena J.R. Levenback C. Mansfield P. Liu J. Angiosarcoma of the spleen clinically presenting as metastatic ovarian cancer. A case report and review of the literature.Ann Diagn Pathol. 2005; 9: 289-292Abstract Full Text Full Text PDF PubMed Scopus (22) Google Scholar but it remains the most common nonhematologic malignancy of the spleen. It is highly aggressive with frequent early metastasis, most commonly to the liver. Neither treatment with chemotherapy nor radiation therapy has been shown to improve outcomes; however, splenectomy can improve survival in localized disease." @default.
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