Recently, de novo truncating variants of SEMA6B uniquely identified in the last exon were reported in five unrelated individuals affected with myoclonic epilepsy, in which the protein truncation in the last exon was found to cause defective development of brain neurons and enhanced seizure behavior in the zebrafish model (Hamanaka et al., 2020). This evidence concerns the gene SEMA6B and myoclonic epilepsy.