A Case of Intralymphatic Histiocytosis Associated with Breast Cancer / 대한피부과학회지

Intralymphatic histiocytosis (IH) is a rare disease with variable and nonspecific clinical features, but with characteristic histopathological findings. Histopathologically, dilated lymphatic vessels containing aggregates of mononuclear cells are observed near the reticular dermis. IH is often associated with other diseases, such as rheumatoid arthritis, osteoarthritis, joint metal implantation, and with breast cancer where it occurs over the mastectomy scar. Although the pathogenesis of IH is not defined, some authors have suggested that lymphatic vessel engorgement by histiocytes migrating from neighboring inflammatory reactions may play a key pathogenic role. We present a case of IH associated with breast cancer. A 50-year-old female developed multiple tender erythematous plaques on both breasts and the right shoulder 3 years after bilateral mastectomy. To our knowledge, this is the first case of IH in association with breast cancer in the Korean dermatologic literature.

A Case of Intralymphatic Histiocytosis after Orthopedic Metal Implants / 대한피부과학회지

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.

A Case of Scar-like Intralymphatic Histiocytosis / 대한피부과학회지

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.

Intralymphatic Histiocytosis: A Rare Self-limited Disease / 대한피부과학회지

There have been a small number of recent case reports of patients with erythematous patches around the joints, which histopathologically showed histiocytic aggregations in the dermal vessels. Intralymphatic histiocytosis (ILH) is a rare group of skin diseases that are characterized by the proliferation of histiocytes in a lymphatic vessel lumen, and this is thought to arise as a benign reaction to certain stimuli such as rheumatoid arthritis. The pathogenesis of this intralymphatic proliferation of histiocytes and the reasons they commonly present on the arms are still unknown. We report on a case of ILH with arthritis in a 68-year old female who had no underlying disease, and the ILH presented as irregular erythematous patches on the left antecubital area and these patches demonstrated the distinctive histopathological features of intralymphatic histiocytosis.