The purpose of this workshop is to stimulate discussions among experts to identify systematic experimental approaches to gain comprehensive insights into psychiatric disease mechanisms based on human genetic findings. This Genes to Biology (G2B) framework is envisioned as a tiered approach from broad, unbiased high-throughput screens to deep, targeted low-throughput investigations. Given that the effects of coding mutations are more readily interpretable, this workshop focuses on identifying current or emerging scalable technologies for surveying the biological impact of all risk alleles in genes with an increased burden of damaging mutations in neurodevelopmental and psychiatric disorders. The hope is to develop a systematic framework for gaining a meaningful biological understanding of the genetic risk underlying psychiatric disorders and make much-needed headway into identifying disease mechanisms.