Multicentric glioblastoma is a uncommon brain malignant tumour.We report the case of a 43-years-old woman, born in Ukraine and living in Italy, who manifested an initial isolated epileptic seizure and subsequent atypical psychiatric symptoms. Clinical neurological examination, Brain Computed Tomography and standard EEG examinations were negative at the moment of admission. A month later, she presented apathy, apraxia, psychomotor slowdown and expressive aphasia. A Magnetic Resonance Imaging examination showed a bi-frontal lesion. The patient underwent to two neurosurgical removals of the lesions: histological examination demonstrated the presence of a grade IV glioblastoma.Clinical onset, diagnostic and therapeutic problems are discussed.In case of atypical psychiatric presentation, it should be taken into consideration neoplastic, inflammatory or infective causes. Despite the absence of focal neurological signs and basal CT scan and EEG alterations, complementary imaging examinations, such as MRI and contrast enhancement CT, are necessary, especially when the conditions become quickly worse.