We report a case of xanthelasma palpebrarum (XP) with bilateral extensive and disfiguring involvement of all four eyelids. The patient was a 28-year-old woman and noticed this condition eight years ago. Physical examination showed yellowish flat topped bilateral extensive plaques around the eyes. Laboratory result was remarkable for a decrease of a serum high density lipoprotein cholesterol (HDL-C). Other laboratory findings were within normal limits. Histopathologic findings showed diffuse infiltrations of foamy cells, histiocytes and foreign body giant cells in the dermis. Electronmicroscopic examination showed that the dermal infiltrate was composed of lipid laden histoncytes showing multiple villi which indicated an activated state of histioncyte.

Bilateral and extensive XP is very rare. Only two cases have been reported in the literatures as far as we know.