The anticonvulsant action of CBD in a mouse model of Dravet syndrome was attributed to antagonism of GPR55, where CBD and the selective GPR55 antagonist CID16020064 [18] similarly restored loss of inhibitory neurotransmission in the dentate gyrus and CID16020064 blocked the effects of CBD [3]. Here, GPR55 is linked to encephalopathy, progressive, early-onset, with brain edema and/or leukoencephalopathy.