To investigate the role of RECQL4 in gliomas/neurofibromatosis type 1 associated neoplasms and susceptibility to treatment, we evaluated the prevalence of RECQL4 genetic alterations in a large cohort of patients with nervous system tumors, assessed the expression of the protein in TMAs, and correlated with clinical, pathological, and molecular aspects, and finally, we assessed the impact of RECQL4-knockdown in survival and vulnerability to ATR-kinase inhibitors in related cell lines (Figure S7). This evidence concerns the gene RECQL4 and central nervous system cancer.