We describe a case of recurrent metastatic malignant ameloblastoma to the lungs with hypercalcaemia in a 47-year-old man. The first lung metastasis was resected nine years after the initial primary, and the tumour recurred with extensive pulmonary metastases 21 years after the primary tumour was resected. This case presented with malignancy-associated hypercalcaemia, likely due to paraneoplastic syndrome, which is exceedingly unusual in association with malignant ameloblastoma. He was successfully treated with carboplatin/paclitaxel and showed the longest survival and stable disease, from the diagnosis of recurrent metastasis, recorded as a case report. This regimen is reasonably well tolerated and can be repeated safely.
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