Central giant cell granuloma is a benign lesion of the jaws which is sometimes aggressive locally. The most common treatment is curettage,which has a high recurrence rate, particularly in more aggressive lesions. Other treatments such as interferon (IFN) and calcitonin have been described. We report a patient with Stickler syndrome and an aggressive central giant cell granuloma in the mandible. Initial treatment with calcitonin was not successful. A combination of IFN and imatinib, given for 9 months, initiated regression of the lesion that continued after treatment had ceased.