GPHN and hyperekplexia: Since artemisinins have been shown to be capable of crossing the blood brain barrier (Davis et al., 2003) and as dysfunctions in gephyrin-mediated neurotransmission have been implicated in severe neurological disorders such as Alzheimer’s disease, autism, schizophrenia, epilepsy and also in hyperekplexia (Agarwal et al., 2008; Fang et al., 2011; Hales et al., 2013; Dejanovic et al., 2014, 2015), the gephyrin-artemisinin co-crystal structures may serve as a starting point for future drug development efforts against these disorders.