iPSCs offer a paradigm shift in central nervous system (CNS) disease modeling. iPSCs can be reprogrammed from a variety of tissue sources and differentiated into many different cell types that would be useful for modeling disease aspects in the CNS. For instance, differentiation into midbrain dopaminergic neurons would provide a cell type applicable for modeling Parkinson’s disease. These disease models and neurodevelopmental processes can be developed into screening assays to interrogate further for drug screening and gene profiling studies. These screening applications can be aided further by the development of iPSC panels of controls, patient-specific lines, isogenic lines, engineered lines, and reporter lines. Abbreviations: iPSC, induced pluripotent stem cell; NSC, neural stem cell.