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Erdheim-Chester disease is a rare non-Langerhans-cell histiocytosis involving multiple organs. On histological evaluation, lipid-laden histiocyte aggregates in Erdheim-Chester disease is detected, but fat tissue in affected organs is not noted grossly on computed tomography. A 40-year-old man presented with bilateral perirenal masses containing fat tissue. He was diagnosed as perirenal involvement of Erdheim-Chester disease. This report describes a case of Erdheim-Chester disease with perirenal involvement that demonstrates unusual features.