Craniopharyngiomas are a rare and exceedingly difficult-to-treat type of brain tumour that can be further divided into papillary and adamantinomatous subtypes. A whole-exome sequencing study of patient tumour samples identified a single driver mutation in each subtype: a BRAFV600E mutation for papillary craniopharyngiomas and a β-catenin mutation for adamantinomatous craniopharyngiomas. These mutations are mutually exclusive and clonal, and they can be used to classify these tumour types in patients. Furthermore, there are currently targeted therapies for the BRAFV600E mutation that could be used to treat papillary craniopharyngiomas.