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Seizure is a prevalent symptom and is an important neurological complaint in the emergency department. Patients with first-time seizures require a thorough evaluation to determine the possible etiologies and identify any causative pathology. Further, neuroimaging studies are vital to identifying the structural culprits. We report the case of a 35-year-old man who was brought to the emergency department with abnormal repetitive shaking movements that were witnessed by his spouse. Before the event, he became dizzy and fell to the ground. During the episode, he was not aware of his surrounding. He developed uprolling of his eyes and had frothy secretions from the mouth. On physical examination, the patient was drowsy but fully oriented. There were no signs of focal neurological deficit. Routine laboratory investigations, including hematological and biochemical profiles, yielded normal results. He was referred to undergo magnetic resonance imaging of the brain. The scan demonstrated the presence of a well-circumscribed lesion in the left Sylvian fissure with high signal intensity on T1- and T2-weighted image with suppression on the fat-suppressed sequence and no post-contrast enhancement. The radiological impression was of Sylvian fissure lipoma. The lesion was successfully resected surgically and the patient had an uneventful recovery with no complaints at the follow-up visits. Sylvian fissure lipoma is among the rarest locations of intracranial lipoma. Despite this, physicians should remember this lesion when they encounter a brain lesion with high signal intensity on T1- and T2-weighted images. While the majority of cases are incidental, an intracranial lipoma can be an etiology of first-time seizures in adults., Competing Interests: The authors have declared that no competing interests exist., (Copyright © 2022, Ghazwani et al.)