Pretibial myxedema is a rare manifestation of Graves disease and is generally associated with ophthalmopathy. Steroids, immunoglobulin, or rituximab can have some therapeutic effect but not curative. Here we reported a 45-year-old man with history of hyperthyroidism for 20 years who developed severe bilateral pretibial myxedema. Three months after the treatment with I for his Graves disease, his pretibial myxedema was significantly improved.