In vitro differentiation of neural precursor cells/neural stem cells (NSC) into different brain cell types is utilized as an acceptable model system to mimic the in vivo neural development [31-36]. Previously, we used a similar in vitro NSC differentiation system to report the first preclinical MECP2 isoform-specific gene therapy vectors, for future gene therapy applications in Rett syndrome [35]. Further, we introduced differentiating NSC as a suitable in vitro model to study the expression and function of developmentally important genes such as Meis1 in neural development [37]. In the current study, we used this previously characterized system to study the expression and regulation of Mecp2 isoforms during NSC differentiation.