Epithelioid hemangioma (EHE) is a rarely malignant tumor originating from the vascular endothelium. Morphological manifestations and immunohistochemical staining results are key to diagnosing EHE. Herein, we present a case of cutaneous involment in a recurrence of EHE. A 75-year-old woman presented with a month-long history of persistent erythematous plaque and pain in the left shoulder. Skin biopsy specimen revealed epithelioid tumor cells containing abundant eosinophilic cytoplasm. Immunohistochemical staining confirmed a recurrence of EHE involving the skin. Subsequently, recombinant human endostatin was administered. At the 10-day follow-up, the cutaneous plaque had improved, and the pain had resolved. When presenting as a local painful erythematous plaque, the possibility of skin involvement in a malignant tumor should be considered. Early diagnosis and early systemic therapy have an important impact on the overall survival of patients with EHE.
