Cilengitide is a novel cyclic peptide that demonstrates anti-integrin activity. In laboratory studies, cilengitide binds with high affinity to the intergrins αvβ3 and αvβ5. It blocks proliferation of cells expressing this class of integrins and results in antiangiogenic and antitumor activity in animal models. Cilengitide has found a niche for clinical development in the population of patients affected by malignant glioma. The clinical evaluation has thus far found an overall high safety profile with limited toxicity and improved clinical outcomes in both recurrent and newly diagnosed glioma patients. This review intends to provide an overview of the preclinical development, summary of clinical trials to date, and update on ongoing and active therapeutic studies.