Application of CRISPR/Cas9 to the study of brain development and neuropsychiatric disease

•CRISPR/Cas9 techniques to study neurodevelopment.•CRISPR/Cas9 applications to hiPSC-based models of neuropsychiatric disease.•The potential for CRISPR/Cas9 approaches to advance our understanding of psychiatric illnesses.

CRISPR/Cas9 technology has transformed our ability to manipulate the genome and epigenome, from efficient genomic editing to targeted localization of effectors to specific loci. Through the manipulation of DNA- and histone-modifying enzyme activities, activation or repression of gene expression, and targeting of transcriptional regulators, the role of gene-regulatory and epigenetic pathways in basic biology and disease processes can be directly queried. Here, we discuss emerging CRISPR-based methodologies, with specific consideration of neurobiological applications of human induced pluripotent stem cell (hiPSC)-based models.