BackgroundCentral giant cell lesion is a non-neoplastic proliferation, usually asymptomatic, of unknown etiology. The purpose of this case report is to report the diagnosis and the treatment of a recurrent central giant cell lesion in the maxilla.Case presentationA 31-year-old Brazilian woman presented to our Surgery Service for evaluation of a cystic lesion in her teeth 13 and 15, although she had previously received endodontic treatment for her teeth 13 and 15 without regression of the lesion. On clinical examination, an increase and painless swelling was observed in her right jaw. An excisional biopsy of the lesion was performed under general anesthesia; the material was sent for pathological examination and a diagnosis compatible with central giant cell lesion was made. She presented again, 10 months after the removal of the lesion, with a recurrent lesion that surrounded her incisors, canine, and right premolar. We suggested that she underwent treatment with intralesional corticosteroids injection. The lesion was significantly reduced and the remainder of the lesion was enucleated. She is monitored at 3-month intervals; at 6 months postoperatively there has been no recurrence.ConclusionsCentral giant cell lesion can have a high degree of invasiveness, which increases the importance of early diagnosis. Combination therapies can provide a favorable prognosis. Periodic monitoring is recommended, thus avoiding the chance of a relapse.