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A case of desmoplastic ameloblastoma recurring within 2 months of curettage is presented. This tumour appeared in the premolar region of the left maxilla with involvement of the antrum. The 24-year-old female patient was initially treated by curettage with wide surgical margins. Later, partial maxillectomy was carried out followed immediately by iliac bone graft. The case was followed with periodic plain radiography and computed tomography. The presence of a pulpally infected premolar and the atypical radiographic appearance obscured the disease. The biologic profile of this tumour is not fully understood because of the limited number of reported cases, coupled with inadequate long-term follow-up. A review of the lesion with emphasis on the pathogenesis of recurrence is discussed.