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Osteoblastoma is a benign bone tumor with predominance in those < 20 years of age and rarely occurs in the maxillofacial region. The identification of FOSgene rearrangements in osteoblastomas has been reported, however, a small subset of osteoblastomas do not show FOSgene rearrangements. There are no reports discussing the relationship between age and FOSgene rearrangement in osteoblastomas. We report the case of a 66-year-old female patient with histological features of osteoblastoma in the mandible without FOSgene rearrangement. According to a review of previously reported cases of osteoblastoma, FOSgene rearrangement is significantly less common in patients > 40 years of age (P = 0.041). Osteoblastomas occurring in older adult patients may exhibit pathogeneses different from those of young patients. Further data, including the relationship between age and FOSgene rearrangement, is necessary to clarify the pathogenesis of osteoblastoma.