Fragile X Syndrome (FXS), the most common hereditary cause of intellectual disability and autism spectrum disorder, is caused by the lack of expression of the Fragile X messenger ribonucleoprotein 1 (FMRP). FMRP is a widely expressed RNA-binding protein that plays a pivotal role in regulating protein synthesis. The absence of its expression has been linked to abnormal synaptic transmission and improper dendritic spines morphogenesis, which are tough to underlies the bulk of the cognitive and behavioral defects exhibited by FXS patients. However, our current understanding of FXS physiopathology remains limited since most research investigations have been conducted with animal models. To overcome this issue, we studied FXS etiology using induced pluripotent stem cells (iPSC) derived from FXS patients. We investigated the neurodevelopmental characteristics of the disease by i) assessing the commitment of iPSC towards neuroectoderm in absence of FMRP expression and by ii) characterizing neural progenitor cells (NPC) proliferation and differentiation. We showed that FXS-derived NPC exhibited dysregulated cell signaling, protein synthesis, proliferation, cell cycle progression and cell fate upon differentiation. Multi-omics analyses of FXS NPC and neurons revealed key molecular insights into how the loss of FMRP expression leads to these phenotypes. We seek to further investigate the potential contribution of these mechanisms to FXS pathophysiology and evaluate their modulation as a potential therapeutic avenue for FXS.