Anastomosing hemangioma: report of two renal cases and analysis of the literature
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CASE REPORT
Open Access
Anastomosing hemangioma: report of two renal cases and analysis of the literature Marina Perdiki1, Galateia Datseri2, George Liapis1, Nikolaos Chondros3, Ioannis Anastasiou4, Maria Tzardi2, Johanna K. Delladetsima1 and Elias Drakos2*
Abstract Background: Anastomosing hemangioma (AH) is a very rare vascular tumor mimicking angiosarcoma, predominately observed in kidney and less frequently in other organs. We present two new renal cases of AH at opposite ends of the clinical presentation spectrum, provide review of the literature and compare the epidemiological, clinical and pathological profiles of renal and non-renal cases. Case presentation: The first occurred in a 64-year-old woman presented with back pain and the second, a multifocal lesion, in a 47-year-old man with end stage renal disease (ESRD). Histology disclosed a vascular tumor with striking anastomosing pattern, minimal nuclear atypia and locally infiltrative pattern, mimicking superficially angiosarcoma. Extramedullary hematopoiesis, extensive perirenal fat entrapment and increased number of mast cells were additional features in the second lesion. Both patients are well, without disease, 25 and 14 months after diagnosis. Conclusion: Comprehensive review and analysis of the published literature show that the growing number of non-renal AHs exhibits similar epidemiologic, clinical, biologic and histologic characteristics with renal AHs and most mild differences vanish after exclusion of cases associated with ESRD. Better understanding of AH pathogenesis will contribute to optimal treatment choices. Keywords: Anastomosing, Hemangioma, Renal, Non-renal, Review
Background In 2009, Montgomery and Epstein described 6 peculiar vascular lesions, 4 in the kidney or perirenal tissue and 2 in the testis, anatomical sites which rarely harbor angiomatous tumors, with histologic features mimicking angiosarcoma and evidence of benign behavior [1]. Because they consisted of capillary-size vessels with prominent anastomosing pattern, the term “anastomosing hemangioma (AH) of the genitourinary tract” was proposed. Since then, more than 50 renal cases, more frequently encountered in the context of end-stage renal disease (ESRD), including bilateral and multifocal lesions and approximately a similar number of extra-renal cases involving various internal organs, bone and soft tissue have been reported, mostly as individual cases or in small series [2–21].
Most lesions are accidental findings, frequently discovered during imaging studies for other tumors in the same, or other organs. All lesions are characterized histologically by anastomosing sinusoidal-like spaces, lined by a single file of CD31/CD34+ and D2-40- endothelial cells, some with hobnail morphology, supported by pericytes, accompanied by mild chronic inflammatory cells, without plasma cells and frequently associated with features of extra-medullary hematopoiesis [1–21]. They are also negative for the presence of human herpes virus 8 (HHV8). Occasionally, periodic acid–Schiff, diastas
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