Recovery of nonketotic hyperglycaemic hemichorea –hemiballismus due to acute ischemic stroke in the contralateral supplementary motor area: a case report and literature review

Introduction:
There remains uncertainty about the mechanism and specific location of the relative cortex with nonketotic hyperglycaemic hemichorea–hemiballismus (HC–HB). This paper aims to analyse the relationship between the disappearance of HC–HB and the supplementary motor area (SMA) infarction in a patient who recovered following an acute ischemic stroke.

Case presentation:
An 83-year-old female patient with diabetes mellitus presenting with severe and refractory involuntary movement after hypoglycaemic therapy was referred to an outpatient neurosurgery department for further intervention. Laboratory, magnetic resonance imaging (MRI) and computed tomography (CT) neuroimaging and physical examinations were performed. After a diagnosis of HC–HB was confirmed, the patient received hypoglycaemic therapy and haloperidol; however, there was no significant improvement. Brain MRI T1-weighted images and CT scans showed high signal intensity involving the bilateral putamen nucleus. CT perfusion and CT angiography showed a hypo-perfusion in the SMA of the right hemisphere without significant vascular occlusion. Then, aspirin and clopidogrel were administered, and the patient’s left leg presented slight involuntary movement three days later. Interestingly, her involuntary movement disappeared again on the second day after the discontinuation of antiplatelet therapy. She was discharged three days later, and her symptoms did not recur during a follow-up for three months.

Conclusions:
The SMA dysfunction caused by the acute infarction could terminate or reset the pathological neural path-way of nonketotic hyperglycaemic HC–HB and contribute to the disappearance of the involuntary movement on the contralateral side. The SMA may be a selective intervention target for patients with refractory nonketotic hyperglycaemic HC–HB.