In the present study, a combination of affinity purification mass spectrometry (AP-MS) coupled with crosslinking of FMRP N-tat was performed to unveil the FMRP N-tat interactome in tsA-201 FMR1 knockout (KO) cells and iPSC-derived neurons of patient with FXS. This evidence concerns the gene FMR1 and fragile X syndrome.