摘要:SummaryGlioblastoma (GBM) is the most aggressive primary brain tumor characterized by infiltrative growth of malignant glioma cells into the surrounding brain parenchyma. In this study, our analysis of GBM patient cohorts revealed a significantly higher expression ofGlycosyltransferase8domain containing 1(GLT8D1) compared to normal brain tissue and could be associated with impaired patient survival. Increasedin vitroexpression ofGLT8D1significantly enhanced migration of two different sphere-forming GBM cell lines. Byin silicoanalysis we predicted the 3D-structure as well as the active site residues of GLT8D1. The introduction of point mutations in the predicted active site reduced its glycosyltransferase activityin vitroand consequently impaired GBM tumor cell migration. Examination of GLT8D1 interaction partners by LC-MS/MS implied proteins associated with cytoskeleton and intracellular transport as potential substrates. In conclusion, we demonstrated that the enzymatic activity of glycosyltransferase GLT8D1 promotes GBM cell migration.Graphical abstractDisplay OmittedHighlights•The glycosyltransferase GLT8D1 is enriched in GBM tissue and cells•In silicoanalysis predicts the 3D structure and the active site of GLT8D1•Enzymatically active GLT8D1 promotes GBM migration•Manipulation of GLT8D1 enzymatic activity decreases GBM migration