By using OPC-specific NG2-Cre or NG2-CreERT transgenic mouse lines, we and others have provided convincing evidence that OPCs, after acquiring Trp53 and NF1 mutations, can be directly transformed into malignant gliomas resembling the proneural subtype of GBM, whenever the mutations were introduced in early or adult stage (Liu et al., 2011; Galvao et al., 2014; Alcantara Llaguno et al., 2015). The gene discussed is CSPG4; the disease is malignant glioma.