BACKGROUND:According to the literature, generalized seizure as a presenting sign of stroke is rare, and in the reported cases it was accompanied by a focal neurological deficit. Presentation of a small ischemic atherothrombotic brain infarction with convulsive generalized seizure is very rare. METHODS:We reported a patient with acute small ischemic atherothrombotic infarction associated with an episode of generalized tonic-clonic seizure, a rare clinical manifestation in this type of stroke. The patient was treated with anti-epileptic therapy after admission. RESULTS:The patient was discharged with oral administration of phenytoin 100 mg TDS, aspirin 80 mg daily, and atorvastatin 40 mg daily. CONCLUSION:Small ischemic atherothrombotic infarction can present only with a seizure without any focal neurological deficit.