Schizophrenia (SCZD) is a severe neurological disorder with a global prevalence of 1%, characterized by a strong genetic component with an estimated heritability of 80–85%. Despite extensive research, the specific cell types affected and the molecular mechanisms underlying the disease remain unclear. In this study, researchers reprogrammed fibroblasts from SCZD patients into human induced pluripotent stem cells (hiPSCs) and differentiated them into neurons. SCZD hiPSC neurons exhibited reduced neuronal connectivity, decreased neurite number, and lower levels of PSD95 and glutamate receptor expression. Gene expression profiles revealed altered activity in the cAMP and WNT signaling pathways. Treatment with the antipsychotic Loxapine improved these phenotypes, suggesting that hiPSC neurons can model SCZD. The study identified 596 genes with significant expression changes, 25% of which have been previously linked to SCZD. These genes are involved in pathways such as glutamate, cAMP, and WNT signaling, which are crucial for synaptic refinement and long-term potentiation. The findings highlight the importance of studying gene expression in cell types relevant to the disease. The study also identified copy number variants (CNVs) associated with SCZD, but no CNVs were found at known SCZD-associated regions. The results suggest that multiple gene misfunctions may disrupt key pathways in SCZD, and that as more SCZD cases are studied using hiPSCs, the number of consistently affected genes may decrease, while the diversity of affected pathways may increase. The study provides valuable insights into the cellular and molecular mechanisms of SCZD and highlights the potential of hiPSCs as a model for studying psychiatric disorders.Schizophrenia (SCZD) is a severe neurological disorder with a global prevalence of 1%, characterized by a strong genetic component with an estimated heritability of 80–85%. Despite extensive research, the specific cell types affected and the molecular mechanisms underlying the disease remain unclear. In this study, researchers reprogrammed fibroblasts from SCZD patients into human induced pluripotent stem cells (hiPSCs) and differentiated them into neurons. SCZD hiPSC neurons exhibited reduced neuronal connectivity, decreased neurite number, and lower levels of PSD95 and glutamate receptor expression. Gene expression profiles revealed altered activity in the cAMP and WNT signaling pathways. Treatment with the antipsychotic Loxapine improved these phenotypes, suggesting that hiPSC neurons can model SCZD. The study identified 596 genes with significant expression changes, 25% of which have been previously linked to SCZD. These genes are involved in pathways such as glutamate, cAMP, and WNT signaling, which are crucial for synaptic refinement and long-term potentiation. The findings highlight the importance of studying gene expression in cell types relevant to the disease. The study also identified copy number variants (CNVs) associated with SCZD, but no CNVs were found at known SCZD-associated regions. The results suggest that multiple gene misfunctions may disrupt key pathways in SCZD, and that as more SCZD cases are studied using hiPSCs, the number of consistently affected genes may decrease, while the diversity of affected pathways may increase. The study provides valuable insights into the cellular and molecular mechanisms of SCZD and highlights the potential of hiPSCs as a model for studying psychiatric disorders.