Asymmetric lenticulostriate arteries in patients with moyamoya disease presenting with movement disorder: three new cases

ABSTRACT

Objective

Unilateral movement disorder associated with moyamoya disease is a rare finding and the mechanism remains to be fully elucidated. Theories postulated include contralateral cerebral ischemic or hemorrhagic lesions, and/or hypoperfusion. However, few studies have reported such patients without contralateral lesions nor hypoperfusion. This study aimed to explore the potential mechanism of those who had neither contralateral cerebral lesions nor hypoperfusion.

Methods

Neuroradiological features of lenticulostriate arteries in three cases with unilateral movement disorder associated with moyamoya disease who had neither contralateral lesions nor hypoperfusion were mainly analyzed.

Results

Angiography and 3 T black-blood T1-weighted intracranial vessel wall imaging both demonstrated a significant asymmetry between bilateral lenticulostriate arteries qualitatively and quantitatively on admission. After one-year follow-up, two patients’ vessel wall imaging indicated that the asymmetry diminished, and the symptoms spontaneously resolved.

Conclusion

This report demonstrated that patients with moyamoya disease with unilateral movement disorder who had neither contralateral lesions nor hypoperfusion may be related to the asymmetry between bilateral lenticulostriate arteries through basal ganglia.

KEYWORDS:

• Moyamoya disease
• movement disorder
• vessel wall imaging
• lenticulostriate artery
• magnetic resonance imaging

Acknowledgments

All authors thank the family for participating in the study.

Author contribution

JX and SL contributed equally to the collection of the patients’ clinical and neuroradiological data, wrote the initial draft of the report, revised and edited the manuscript. XJ, QZ proposed and supervised the case study and edited the manuscript. GBR, WZ, YD were involved in treatment planning and edited and revised the manuscript.

Disclosure statement

The authors declare that the research was conducted in the absence of any commercial or financial relationships that could be construed as a potential conflict of interest.

Additional information

Funding

This project was supported by the National Natural Science Foundation of China (No. 81801313), Beijing Municipal Administration of Hospitals’ Youth Program (No. QML 20180801), National Key R&D Program of China (2017YFC1308405) and Cheung Kong Scholars Program (T2014251)

Notes on contributors

Jiali Xu

Jiali Xu is a MD candidate at Capital Medical University in the Department of Neurology, and mainly researches on cerebrovascular diseases.

Sijie Li

Sijie Li is a PhD researcher at Beijing Key Laboratory of Hypoxic Conditioning Translational Medicine, Xuanwu Hospital.

Gary B. Rajah

Gary B. Rajah is a cheif physician at Department of Neurosurgery of Wayne State University and majors in MMD.

Wenbo Zhao

WenBo Zhao is a chief physician Capital Medical University in the Department of Neurology, majors in cerebral ischemic preconditioning, acute ischemic stroke, hemorrhagic stroke.

Changhong Ren

Changhong Ren is a researcher at Beijing Key Laboratory of Hypoxic Conditioning Translational Medicine of Xuanwu Hospital,specializes in cerebral ischemic preconditioning, hypothermic neuroprotection.

Yuchuan Ding

Yuchuan Ding is at the forefront of investigations into the mechanisms underlying ethanol and normobaric oxygenation-induced brain protection following cerebral ischemia. Who do manuscript writing and editing portions.

Qian Zhang

Zhang Qian is a chief physician at Capital Medical University in the Department of Neurology, she specializes in intracranial atherosclerotic stenosis, moyamoya disease.

Xunming Ji

Xunming Ji is a professor at Capital Medical University in the Department of Neurology. He also serves as Vice President of Xuanwu Hospital at Capital Medical University. He specializes in cerebral ischemic preconditioning, hypothermic neuroprotection, neurosurgical treatment of intracranial venous sinus disease, internal jugular venous disease and moyamoya disease.