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Effects of bilateral pallidal deep brain stimulation for chorea after pulmonary thromboendarterectomy with deep hypothermia and circulatory arrest: case report

K. Aoyagi, Y. Higuchi, Y. Okahara, Y. Yamanaka, T. Yamamoto, S. Hirano, Y. Iwadate (Ichihara, Japan)

Meeting: 2017 International Congress

Abstract Number: 308

Keywords: Chorea (also see specific diagnoses, Deep brain stimulation (DBS), etc): Treatment, Globus pallidus, Huntingtons disease

Session Information

Date: Monday, June 5, 2017

Session Title: Surgical Therapy: Other Movement Disorders

Session Time: 1:45pm-3:15pm

Location: Exhibit Hall C

Objective: We report bilateral pallidal deep brain stimulation (DBS) improved chorea after pulmonary thromboendarterectomy (PTE) with deep hypothermia and circulatory arrest.

Background: Chronic thromboembolic pulmonary hypertension (CTEPH) and chronic pulmonary thromboembolism (CPTE) are forms of pulmonary hypertension caused by non-resolving thromboembolisms of the pulmonary arteries. PTE is an effective surgical treatment option for CTEPH and CPTE. During PTE, deep hypothermia and circulatory arrest are adopted for the protection of brain. Choreoathetosis after PTE had been reported previously. Transient hypoperfusion in basal ganglia may be involved in the pathophysiology. An increasing knowledge of functional neurosurgery allowed DBS to be a potential treatment of rare hyperkinetic movement disorders. However, we found no report demonstrating the efficacy of globus pallidus interna (GPi) -DBS for chorea after PTE.

Methods: A 41-year-old man was admitted with complaints of worsening exertional dyspnea and cough for 6 years. He was diagnosed with CPTE, and underwent PTE with deep hypothermia and circulatory arrest for 85 minutes. Five days after the operation, chorea emerged in the lower extremities. Magnetic resonance imaging (MRI) of the brain showed bilateral T2 elongation in bilateral caudate nuclei 17 days after the surgery. Despite treatment with medications, only clonazepam slightly suppressed choreatic movements. The patient was referred to our hospital due to disabling chorea 16 years after PTE. Neurological examination revealed choreatic movements in the four extremities at rest and in walking and no cognitive impairment. Brain MR images indicated atrophy in the bilateral head of caudate nuclei. Unified Huntington’s disease rating scale (UHDRS) motor assessment was 27.

Results: He underwent GPi-DBS. Intraoperative microelectrode recordings demonstrated low activities in the GPi. Continuous GPi stimulation almost diminished his hyperkinetic involuntary movement without any remarkable side effects. UHDRS motor assessment decreased to 12.

Conclusions: GPi-DBS may be a treatment option for sustained choreatic movements after PTE with deep hypothermia and circulatory arrest.

To cite this abstract in AMA style:

K. Aoyagi, Y. Higuchi, Y. Okahara, Y. Yamanaka, T. Yamamoto, S. Hirano, Y. Iwadate. Effects of bilateral pallidal deep brain stimulation for chorea after pulmonary thromboendarterectomy with deep hypothermia and circulatory arrest: case report [abstract]. Mov Disord. 2017; 32 (suppl 2). https://www.mdsabstracts.org/abstract/effects-of-bilateral-pallidal-deep-brain-stimulation-for-chorea-after-pulmonary-thromboendarterectomy-with-deep-hypothermia-and-circulatory-arrest-case-report/. Accessed June 15, 2025.
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