In this study, we used CRISPR/Cas9 to knockout AS3MT in the human SH‐SY5Y neuroblastoma cell line to investigate the role of AS3MT in neuronal cell function and explore its effect on cell morphology, AS3MT isoform usage, and the transcriptomic regulation of other genes in a cancer model (Figure 1). Here, AS3MT is linked to neuroblastoma.