Pimozide effectively suppressed USP1 expression, reduced N-Myc levels, and inhibited neuroblastoma cell proliferation.<h4>Conclusion</h4>This study uncovered a novel oncogenic axis in neuroblastoma, where NeuroD1 transcriptionally upregulates USP1, promoting N-Myc stabilization and tumor progression. This evidence concerns the gene NEUROD1 and neuroblastoma.