We present a patient who had a large metastatic pulmonary ameloblastoma resected 25 years after removal of an apparently benign primary ameloblastoma of the jaw. It highlights three areas: problems with the histopathological diagnosis of pulmonary metastases using fine needle aspiration; a noted radiological improvement after a 5-year course of oral cyclophosphamide, in keeping with occasional patients who have responded to chemotherapy; and the technical difficulties of resection of a large pulmonary metastasis, particularly when it is adherent to the mediastinum.
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