An intraosseous lipoma is a rare benign bone lesion that proliferates from mature lipocytes. It occurs most frequently in the lower limb, particularly in the calcaneus. The talus is an unusual location for this rare lesion. A review of the literature produced only two reports with talar intraosseous lipomas under the name of intraosseous lipomatosis, which described multiple lipomas in different areas. We describe a 38-year-old male patient who had an isolated intraosseous lipoma with an osteochondral defect in the talus and was treated with autologous osteochondral graft transplantation by medial malleolar osteotomy. He could walk with full weightbearing without any assistance at the end of 12 months. Intraosseous lipoma localized in the talus may be confused radiologically with other bone lesions, especially with unicameral bone cyst, if it is associated with an osteochondral defect. Autologous osteochondral graft transplantation is a successful treatment method for talar intraosseous lipoma.