Updated project metadata. Disease-modifying therapies remain an important unmet need for neurodegenerative diseases, including Parkinson’s disease (PD). The aim of this study was to investigate the molecular pathways affected by the multi-kinase inhibitor BX795, which is neuroprotective in an induced pluripotent stem cell (iPSC)-based model of familial PD. To this end, we performed proteomics analysis in iPSC-derived neurons from patients bearing the G209A (p.A53T) α-synuclein (αSyn) mutation vis-à-vis control neurons, in the absence or presence of BX795. Comparison between p.A53T versus control neurons in the absence of BX795, revealed differential expression of 640 proteins from which only 67 were down-regulated whilst the rest 573 were up-regulated. This large increase in protein expression was linked by GO enrichment analysis mainly to the biological processes of transcription, translation, protein synthesis and modification. Upon treatment with BX795, the levels of a cohort of 118 proteins, lying mostly within these biological processes and representing approximately 20% of the total dysregulated proteins in p.A53T neurons, were restored. Enrichment analysis using the DAVID and Reactome softwares showed that these proteins are predominantly associated with mRNA metabolism, mRNA transport and translation, protein metabolism and degradation processes. This outcome was specific to p.A53T-neurons as BX795 had no significant effect on the proteome of control neurons.