An integrated in vitro human iPSCs-derived neuron and in vivo animal approach for preclinical screening of anti-seizure compounds

•A human iPSCs-based high-throughput anti-seizure compounds screening platform was built.•The human iPSCs-derived neurons can effectively and efficiently predict positive compounds.•This integrated anti-seizure drug discovering approach combines the advantages of human genetic background, HTS, and animal brain network.•Piperine, magnolol, α-asarone, and osthole can reduce epileptiform activity and seizures.•Natural compounds are promising candidates used for anti-seizure drug discovery.