Recent discoveries of multiple driver mutations open promising perspectives for targeted therapies in meningioma. Nevertheless, iterative recurrences of most aggressive meningiomas as extended skull base meningiomas are not systematically operated and histologically documented. This suggests the interest and the relevance of liquid biopsy in meningiomas. In a proof-of-concept study, we detected the NF2 mutation in 2 of 6 cell-free plasma DNAs and 1 of 1 cerebrospinal fluid (CSF) from high-grade recurrent cases, suggesting that identification of the driver mutation in blood and CSF is today feasible. Liquid biopsy could be an interesting tool to adapt the targeted therapy in meningiomas in the near future.