Hemichorea-hemiballismus (HCHB) is a rare hyperkinetic movement disorder characterized by unilateral, involuntary, and irregular movements. Although HCHB is the most common poststroke movement disorder, its occurrence as a complication of intravenous thrombolysis is extremely uncommon. While previous studies have demonstrated that intravascular interventions and intravenous thrombolysis can effectively alleviate HCHB symptoms in stroke patients, the pathophysiologic mechanisms underlying HCHB induced by reperfusion therapy remain poorly understood. Herein, we report a case of HCHB induced by intravenous thrombolysis in an acute ischemic stroke patient and explore its possible pathophysiologic underpinnings. Through detailed clinical observations, comprehensive neuroimaging analyses, and an extensive literature review, we investigated the relationship between reperfusion therapy and HCHB onset. Our findings suggest that HCHB induced by reperfusion therapy may be associated with increased metabolic activity in the basal ganglia or reperfusion injury following the restoration of cerebral blood flow. This study provides novel insight into this rare complication, raises clinician awareness, and lays the foundation for future research into HCHB's mechanisms and treatment.