Objective: To describe a rare cause of unilateral chorea caused by a structural lesion with few descriptions in literature.

Background: While chorea typically manifests in a generalized manner, involving the entire body, it may also present asymmetrically or unilaterally. Unilateral chorea is often linked to structural abnormalities and can stem from various underlying conditions. Within this case report, we described a rare etiology associated with hemichorea.

Method: We described an isolated case of hemichorea secondary to an enlarged perivascular space. We delineate the clinical features of the hyperkinetic disorder, alongside the results of ancillary investigations. Regarding imaging studies, brain computed tomography (CT) and 3T MRI imaging were conducted.

Results: A 67-year-old male patient was brought to the emergency department by his daughter who noticed that he was experiencing involuntary movements in the right hemibody. The onset of symptoms remained uncertain as the patient had not initially perceived the movements. His previous medical history was remarkable for multiple myeloma, exhibiting a complete response to chemotherapy and autologous bone marrow transplant performed in 2022, as well as chronic obstructive pulmonary disease and hypertension. Neurological examination revealed choreiform movements predominantly affecting the distal aspect of the right hemibody, including ipsilateral hemiface. Brain CT and angio-CT were conducted to investigate possible underlying causes, yielding no significant findings apart from an oval hypodensity in the left nucleocapsular region. Subsequent 3T brain MRI revealed an enlarged perivascular space in the left nucleocapsular and sublenticular regions, measuring 1.6×1.6 centimeters. All laboratory tests, including metabolic, hematological, and rheumatological profiles, showed no alterations. Further investigation failed to identify additional conditions justifying the observed symptoms.

Conclusion: Despite enlarged perivascular spaces are typically asymptomatic, our case highlights that giant perivascular spaces, especially when near the basal ganglia, can manifest as unilateral movement disorders, including chorea. This underscores the importance of considering this rare etiology in the differential diagnosis of such conditions.

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MDS Abstracts - https://www.mdsabstracts.org/abstract/a-rare-case-of-hemicorea-secondary-to-giant-perivascular-space-a-case-report/