TAOK1 is closely associated with neurodevelopment and neurodegeneration. It has been confirmed to be highly expressed in patients with Alzheimer's and Parkinson's disease accompanied by cognitive disorder, and is therefore considered a potential biomarker. In this study, we observed through neuronal morphology and protein expression that TAOK1 overexpression leads to shorter neurite in differentiating SH-SY5Y cells. The protein expression of both α-tubulin and β-tubulin was also decreased, which may be one of the reasons for the observed neurite shortening. Furthermore, we found a significant positive correlation between TAOK1 expression and mental illness-related gene, MAO-A. The mRNA expression of TDP-43, a protein related to neurodegeneration, also significantly increased, with a notable rise in phosphorylated TDP-43 protein expression as well. Therefore, the findings of this study may offer new diagnostic and therapeutic strategies for neurodegenerative and psychiatric disorders.