We previously developed a protocol for the rapid differentiation of PPN-enriched cultures from hiPSCs.17 In this study, we exploited that system for the in-depth characterization of sensory neurons derived from healthy donors (CT) and FRDA patients as well as from FRDA sibling ISO CTs.13–15 We started from the epigenetic analysis of the FXN locus and with the investigation of the transcriptomic and proteomic profile of developing or fully differentiated neurons. The gene discussed is FXN; the disease is Friedreich ataxia.