Intralymphatic atypical T-cell proliferation in a cutaneous hemangioma

We report an unusual case of atypical T-cell proliferation involving the lymphatic vessels within a cutaneous hemangioma from an elderly woman. Despite the blastic morphology, the CD4 restricted phenotype and the very high proliferation index, the clinical presentation (single skin lesion in a healthy woman), the benign clinical course and the absence of T-cell receptor (TCR) clonal rearrangement favored a reactive nature of the process. Since the atypical cells showed an effector/memory-like regulatory T-phenotype (CD45RO+, CD25+ and FOXp3+), expressed the migration-associated molecule CCR7 and were exclusively located within podoplanin+ lymphatic vessels, we speculate that the process might reflect an unusual local immune response, with migration of T-cells to draining lymph nodes.