Background: Ticagrelor is an antiplatelet agent widely used in the treatment of coronary acute syndrome and was recently found to be of interest in secondary prevention of minor stroke and transient ischemic attack. One retrospective study considers ticagrelor as a safe option for secondary prevention after moderate or severe acute ischemic stroke. The eligibility for intravenous thrombolysis in patients pretreated with ticagrelor is controversial. Literature on the safety of intravenous thrombolysis for ischemic stroke in patients taking ticagrelor is limited to eight cases, of which two cases of symptomatic intracranial hemorrhage, one case of asymptomatic intracranial hemorrhage and one case of retropharyngeal hematoma have been reported. Case presentation: We report a 57-year-old patient who was hospitalized for ST-segment elevation myocardial infarction and treated by ticagrelor. Four days later, he presented an anterior circulation ischemic stroke clinically expressed by right hemiparesis with mutism. The patient was successfully treated by intravenous thrombolysis with an improvement of the National Institutes of Health Stroke Scale from 12 to 1 at 24 hours 0 . No complications have been observed post-treatment. Conclusions: This is the second reported case of successful intravenous thrombolysis for an ischemic stroke of the anterior circulation in a patient pretreated with ticagrelor. This case reinforces the urgent need for further investigations on the safety of intravenous thrombolysis in patients pretreated with ticagrelor as the number of such cases is expected to increase in the future.