A case of recurrent ameloblastoma developing in an autogenous iliac bone graft 20 years after radical resection of primary mandibular ameloblastoma is presented. This case showed extensive bony resorption extending throughout the auto-grafted bone. Seeding to the graft bone was suspected as a cause of the recurrence in the iliac bone graft because wide destruction of the graft bone was observed without any prior signs and symptoms of intraoral mucosa. Consequently, extensive resection of bone as well as adjacent soft tissue was performed. Persistent follow-up examination is essential for management of ameloblastoma.