BACKGROUND AND IMPORTANCE: The suprasellar and hypothalamic/chiasmatic regions can harbor a broad range of pathologic conditions, both neoplastic and nonneoplastic; however, malignant gliomas are extremely rare in those regions. CLINICAL PRESENTATIONS: Patient 1 was a 70 year-old man with weight loss and rapidly progressive visual impairment. A mass centered in the hypothalamus was detected on magnetic resonance (MR) imaging. The second patient, a 45 year-old woman, complained of visual symptoms and headaches. MR imaging revealed a combined intra- and suprasellar mass. In both instances, the preoperative differential diagnosis favored craniopharyngioma. Histological examination confirmed the diagnosis of glioblastoma. CONCLUSION: We report two rare adult cases of hypothalamic/chiasmatic glioblastoma. The authors review the literature, highlighting the importance of considering this rare entity in the differential diagnosis of suprasellar and hypothalamic lesions.