Our findings support that in GBM, MTUS1/ATIP1 acts as a TSG, and we believe that a detailed elucidation of the role of MTUS1/ATIP1 in GBM might help to develop new strategies that target MTUS1/ATIP1-associated pathways and that might help to slow down or even block the progression of glioma. Here, MTUS1 is linked to central nervous system cancer.