In BRAFV600E-positive ameloblastomas which were unresectable due to multiple recurrences and lung metastases, monotherapy with BRAFi dabrafenib [19,21] or vemurafenib [20], neoadjuvant treatment with dabrafenib [24,25,26], or dual BRAF/MEK inhibition with dabrafenib and trametinib [18,22,23,26] showed good response without severe toxicity. This evidence concerns the gene BRAF and ameloblastoma.