Abstract
BackgroundThe spectrum of Kaposi's sarcoma (KS) has been expanded to include pre-KS lesions.Case PresentationWe report, for the first time, a case providing direct histological evidence of the development of early (in-situ) KS from mediastinal lymphatic vessels in the setting of chronic lymphedema in an HIV-positive patient. Spindle-shaped and endothelial cells in these early KS-appearing lesions were immunoreactive for HHV8, D2-40 and CD34.ConclusionOur findings suggest that HHV8-infected spindle-shaped cells associated with lymphangiogenesis that evolve into KS lesions, acquire from the outset an aberrant mixed vascular and lymphatic endothelial cell phenotype.
Highlights
The spectrum of Kaposi's sarcoma (KS) has been expanded to include pre-KS lesions.Case Presentation: We report, for the first time, a case providing direct histological evidence of the development of early KS from mediastinal lymphatic vessels in the setting of chronic lymphedema in an HIV-positive patient
The D2-40 antibody is another selective marker of lymphatic endothelium and reacts with KS lesional cells at all stages of progression, supporting the concept that KS originates from a stem cell capable of undergoing lymphatic differentiation [3]
We believe that the findings in this case provide direct morphological evidence of the development of an in-situ form of KS directly from lymphatics in the setting of chronic lymphedema
Summary
Kaposi's sarcoma (KS) is a vascular neoplasm that may involve mucocutaneous and visceral body sites. A 34-year-old homosexual male with acquired immune deficiency syndrome (AIDS)-related KS presented with chylothoraces due to obstruction of his thoracic duct by KS He had extensive cutaneous lesions on the face, forehead, upper torso, mid-abdomen, left arm and left flank. Since placement of the shunt, he developed ascites with subcutaneous extravassation of lymph that was associated with xanthogranulomatous bile lakes (Figure 1) He received diuretic therapy and medium-chain triglyceride dietary supplementation with only temporary improvement of his ascites. He was placed on palliative paclitaxel and died of progressive KS about 18 months later His pleural and lung biopsies showed dilated pleuropulmonary lymphatics (Figure 2) with interstitial pulmonary extravassation of lymph. The biopsy revealed a multifocal increase in spindle-shaped cells with neo-angiogenesis originating from dilated lymphatics, associated with scattered lymphocytes and hemosiderin-laden macrophages, resembling early stage KS (Figure 3). Native blood vessel endothelium was HHV8 and D2-40 negative, and only CD34 positive
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