A Case of Scar-like Intralymphatic Histiocytosis

Choi, Jin Hwa; Kim, Byeong Su; Sohng, Seung Hyun; Shin, Dong Hoon; Choi, Jong Soo; Bae, Young Kyung

Abstract

Intralymphatic histiocytosis (ILH) is a rare, chronic cutaneous condition characterized by the presence of dilated lymphatic vessels containing aggregates of mononuclear histiocytes within lumina. ILH presents with asymptomatic and poorly demarcated, erythematous plaques or livedo reticularis-like lesions, usually located on the extremities. ILH is often associated with rheumatoid arthritis, metal joint implants, and mastectomy scars of breast cancer patients, but its pathogenesis remains uncertain. A 69-year-old woman with osteoarthritis, who had a past history of cancer in the left breast, presented with asymptomatic, linear-shaped, scar-like erythematous plaques on the right elbow for a month. Approximately a month before her visit, she had been treated with an intraarticular corticosteroid injection on the right elbow in a local clinic. Histopathologic findings showed irregularly dilated vessels with intraluminal cells in the superficial and deep dermis. In double immunohistochemical staining with CD68/D2-40, endothelial cells lining the vessels were positive for D2-40 staining, which is a marker for lymphatic endothelial cells, and intraluminal cells were positive for CD68 staining, which is a marker for histiocytes. We present the findings of a patient with a rare condition of intralymphatic histiocytosis with scar-like morphology of the plaques.