101449

Authors

• Koki Onodera, Department of Cerebrovascular Surgery, Saitama Medical University International Medical Center, Hidaka, Japan.
• Hiroki Sato, Department of Cerebrovascular Surgery, Saitama Medical University International Medical Center, Hidaka, Japan. Electronic address: hiro1207@saitama-med.ac.jp.
• Milan Lepic, Department of Cerebrovascular Surgery, Saitama Medical University International Medical Center, Hidaka, Japan; Clinic for Neurosurgery, Faculty of Medicine of the Military Medical Academy, University of Defense, Belgrade, Serbia.
• Kaima Suzuki, Department of Cerebrovascular Surgery, Saitama Medical University International Medical Center, Hidaka, Japan.
• Hidetoshi Ooigawa, Department of Cerebrovascular Surgery, Saitama Medical University International Medical Center, Hidaka, Japan.
• Hiroki Kurita, Department of Cerebrovascular Surgery, Saitama Medical University International Medical Center, Hidaka, Japan.

Document Type

Article

Abstract

BACKGROUND AND IMPORTANCE: Although hypoperfusion of the basal ganglia or the frontal subcortical matter is suspected, the pathology of chorea in moyamoya disease remains unclarified. Herein, we report a case of moyamoya disease presenting with hemichorea and evaluate pre- and postoperative perfusion using single photon emission computed tomography with N-isopropyl-p-I-iodoamphetamine (I-IMP SPECT). CLINICAL PRESENTATION: An 18-year-old woman presented with choreic movement of her left limbs. Magnetic resonance imaging revealed an ivy sign, and I-IMP SPECT demonstrated decreased cerebral blood flow (CBF) and cerebral vascular reserve (CVR) values in the right hemisphere. The patient underwent direct and indirect revascularization surgery to improve cerebral hemodynamic impairment. The choreic movements entirely resolved immediately after surgery. Although CBF and CVR values in the ipsilateral hemisphere demonstrated by quantitative SPECT increased, these did not reach the normal values threshold. CONCLUSION: Choreic movement in moyamoya disease may be related to cerebral hemodynamic impairment. Further studies are required to elucidate its pathophysiological mechanisms.

Keywords

Chorea, Involuntary movement, Moyamoya disease, Revascularization surgery

Medical Subject Headings

Humans; Female; Adolescent; Moyamoya Disease (complications, diagnosis, surgery); Chorea (etiology, surgery); Iofetamine; Tomography, Emission-Computed, Single-Photon (methods); Cerebrovascular Circulation (physiology); Cerebral Revascularization (methods); Iodine Radioisotopes

Publication Date

7-1-2023

Publication Title

Neuro-Chirurgie

E-ISSN

1773-0619

Volume

69

Issue

4

PubMed ID

37178488

Digital Object Identifier (DOI)

10.1016/j.neuchi.2023.101449

Recommended Citation

Onodera, Koki; Sato, Hiroki; Lepic, Milan; Suzuki, Kaima; Ooigawa, Hidetoshi; and Kurita, Hiroki, "101449" (2023). Translational Neuroscience. 2599.
https://scholar.barrowneuro.org/neurobiology/2599