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We present the case of an exceptional lipoma of the left sylvian region associated with saccular aneurysm of middle cerebral artery, in a 50 years old female patient who presented with right partial seizures with secondary generalisation. The sylvian fissure is a rare site of intracranial lipomas. The present case is probably the second in the world literature having association of sylvian fissure lipoma with saccular aneurysm of middle cerebral artery. The clinical presentation and findings of the imaging studies which include tomography of the brain, magnetic resonance imaging of the brain and magnetic resonance angiography of the cerebral vessels are described along with a brief review of the literature.