The authors report the case of a 31-year-old man, who presented an hemorrhagic onset of a cerebral-isolated sarcoidosis, to date never described in the literature. Unusual clinical manifestation, neuroradiological study and direct histological confirmation are presented. Moreover, Gallium-67 Single Photon Emission Computed Tomography (Ga-67 SPECT) contribution is indicated to complete the evaluation of patients with cerebral inflammatory lesions, such as neurosarcoidosis, and in cases of equivocal neuroradiological imaging, to evaluate uptake activity of the inflammatory tissue. The patient did well and reported no further progression of his disease during 7 months of follow-up.