Osteoblastoma is a rare tumor which forms osteoid and bone with highly vascular stroma, account for less than 1 percent of all bone tumors. The lesion had been though to be controlled with curettage and local excision. In recent years, osteoblastomas have been described as aggressive osteoblastoma or malignant osteoblastoma, separated from the innocuous behavior of usual osteoblastoma. It represents the histologic characteristics of the presence of epithelioid osteoblasts, trabecular rather than lace-like osteoid, low mitotic rate with no atypical form with prominent giant cells of osteoclastic type. The differential diagnosis between aggressibe osteoblastoma and osteogenic sarcoma is extremely difficult, but its peculiar histologic pattern and its different clinical and radiologic features and better prognosis are helpful. We report here a case of aggressive osteoblastoma of 17 years old female patient with 3 years and 4 months follow-up period and review the literature.