A Case of Intralymphatic Histiocytosis after Orthopedic Metal Implants

Lee, Dong Yoon; Kim, Jun Ki; Choi, Ki Hwa; Lee, Ji Yeoun; Yoon, Tae Young

Abstract

A 64-year-old female presented with a 1-year history of livedo-like erythematous patches and plaques around the surgical scar on the left knee. Examinations revealed poorly demarcated, erythematous, indurated patches and plaques, with mild tenderness overlying the medial and inferior portion of the surgical scar. Histopathology revealed a mixed dermal infiltrate with glomeruloid intravascular accumulation of histiocytes and neutrophils. Histiocytes were identified by immunostaining for CD68. Immunostains for CD31 and D2-40 confirmed the intra-lymphatic location of the histiocytes. Recently, there have been several case reports of erythematous patches around the joints, which histopathologically show histiocytic aggregations in dermal vessels. Intra-lymphatic histiocytosis is a rare group of skin diseases characterized by the proliferation of histiocytes in the vessel lumen. Although the pathogenesis of intra-lymphatic histiocytosis is unknown, a role for lymphatic stasis secondary to chronic inflammation or surgery has been suggested. We present the case of a 64-year-old female who developed a tender rash on her left knee 1 year after orthopedic metal implants. To our knowledge, this is the first case of intra-lymphatic histiocytosis reported in association with a metal implant in the Korean dermatologic literature.