A 54-year-old woman with a 6-month history of asymptomatic edematous erythema on her right eyelid visited our hospital. Rosacea had been present since the age of 47 years. Histological examination of the lesion revealed many granulomas and dilated vessels from the upper dermis to fat tissue. Within one of the dilated vessels, a massive aggregate composed of mononuclear cells was found. Immunohistochemical staining for CD68, a marker for histiocytes, showed intense staining of most cells in the intraluminal aggregate. Staining with D2-40, a marker for lymphatic endothelial cells, revealed that the vessels were lymphatic in nature. We diagnosed our patient as granulomatous blepharitis with intralymphatic histiocytosis based on the clinical and histopathological findings. The patient was treated with intralesional corticosteroids (triamcinolone acetonide) and oral tranilast administration (300 mg/day), and the swelling of her right eyelid was dramatically reduced. We concluded that this was a rare case in which a patient with granulomatous blepharitis also had intralymphatic histiocytosis.
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