The genetic underpinnings of schizophrenia are multifactorial,41 making it challenging to identify convergent points of common pathways, which is essential for developing novel therapeutic and preventive measures. The cells from schizophrenia patients with the 22q11.2 deletion can serve as an entry point for teasing apart this complexity and resolving the principal pathways shared among ‘general' schizophrenia, because this copy number variant is one of the largest effect-size predisposing factors. In this study, we revealed several characteristic features of hiPSCs and their differentiated cell lineage derived from schizophrenia patients with the 22q11.2 microdeletion.