- Department of Neurosurgery, Kumamoto General Hospital, Yatsushiro, Japan.
- Department of Neurosurgery, Amakusa Medical Center, Amakusa, Japan.
- Division of Speech-Language and Hearing Therapy, Faculty of Health Science, Kumamoto Health Science University, Kita-Ku, Japan.
- Department of Neurosurgery, Kumamoto Neurosurgical Hospital, Chuo-Ku, Kumamoto, Japan.
Department of Neurosurgery, Kumamoto General Hospital, Yatsushiro, Japan.
DOI:10.25259/SNI_171_2021Copyright: © 2021 Surgical Neurology International This is an open-access article distributed under the terms of the Creative Commons Attribution-Non Commercial-Share Alike 4.0 License, which allows others to remix, tweak, and build upon the work non-commercially, as long as the author is credited and the new creations are licensed under the identical terms.
How to cite this article: Hirotaka Inoue1, Rihito Yamamura2, Kazumichi Yamada3, Tadashi Hamasaki1, Nobuhiro Inoue4, Akitake Mukasa1. Hemichorea induced by a sphenoid ridge meningioma. 03-May-2021;12:201
How to cite this URL: Hirotaka Inoue1, Rihito Yamamura2, Kazumichi Yamada3, Tadashi Hamasaki1, Nobuhiro Inoue4, Akitake Mukasa1. Hemichorea induced by a sphenoid ridge meningioma. 03-May-2021;12:201. Available from: https://surgicalneurologyint.com/?post_type=surgicalint_articles&p=10787
Background: Movement disorders are rare in brain tumors. We describe a 45-year-old woman with hemichorea, a concomitant contralateral sphenoid ridge meningioma.
Case Description: The meningioma enlarged as her hemichorea worsened, and after meningioma resection, the hemichorea gradually subsided. N-isopropyl-p-[123I]-iodoamphetamine single-photon emission computed tomography performed preoperatively showed decreased regional cerebral blood flow (CBF) to the basal ganglia circuit ipsilateral to the tumor and, when repeated postoperatively, confirmed improved regional CBF.
Conclusion: We propose that the enlarging sphenoid ridge meningioma had a remote effect on regional CBF and the thalamocortical motor center and that complex changes in the basal ganglia output may have caused the hemichorea.
Keywords: Cerebral blood flow, Chorea, Meningioma, Remote effect
Chorea is defined as a hyperkinetic movement disorder, characterized by involuntary, brief, random, and irregular muscle contractions.[
Our patient was a 45-year-old woman who experienced twitching of the left lip, anarthria, and abnormal movements involving the distal part of her left limb 2 years previously. A diagnosis of chorea was made after consultation with the department of neurology, and the symptoms failed to improve despite administration of clonazepam. A small meningioma arising from the right sphenoid ridge was discovered. The meningioma enlarged over time [
N-isopropyl-p-[123I]-iodoamphetamine single-photon emission computed tomography (123I-IMP SPECT) performed before surgery (upper panels) demonstrates decreased regional cerebral blood flow to the right ventral midbrain, thalamus (arrows), and frontotemporal lobe cortex. SPECT performed 5 months after surgery (lower panels) reveals a slight improvement.
Hemichorea generally occurs as a complication of a vascular malformation, basal ganglia tumor, stroke, metastasis, or metabolic disorders.[
We believe that the meningioma caused hemichorea in our patient for the following reasons. First, the laterality of hemichorea corresponded to the tumor location. Second, the hemichorea worsened when the meningioma increased in size, and subsided after surgery, while the APS worsened throughout this period. Third, after tumor resection, the reduced CBF in the midbrain and thalamus normalized.
Movement disorders are rare clinical features of brain tumors.[
We used the Alexander and Crutcher model to determine the causal association of CBF change and hemichorea.[
Herein, we report a rare case of hemichorea induced by a sphenoid ridge meningioma. The symptoms underwent remission after resection of the tumor. We believe that the growing meningioma had a remote effect, altering the firing pattern of the thalamocortical motor pathways and leading to the manifestation of hemichorea. APS can also cause hemichorea; however, the status of the APS in our patient did not correlate with her clinical course.
The authors confirm that the approval of an Institutional Review Board was not required for this work. Written informed consent for the publication of this report was obtained from the patient, and this report does not identify the individual. We confirm that we have read the journal’s position on issues involved in ethical publication and affirm that this work is consistent with those guidelines.
The authors certify that they have obtained all appropriate patient consent.
There are no conflicts of interest.
1. Alexander GE, Crutcher MD. Functional architecture of basal ganglia circuits: Neural substrates of parallel processing. Trends Neurosci. 1990. 13: 266-71
2. Bhatoe HS. Movement disorders caused by brain tumours. Neurol India. 1999. 47: 40-2
3. Cardoso F, Seppi K, Mair KJ, Wenning GK, Poewe W. Seminar on choreas. Lancet Neurol. 2006. 5: 589-602
4. Cervera R, Piette JC, Font J, Khamashta MA, Shoenfeld Y, Camps MT. Antiphospholipid syndrome: Clinical and immunologic manifestations and patterns of disease expression in a cohort of 1, 000 patients. Arthritis Rheum. 2002. 46: 1019-27
5. Chorobski J. Involuntary movements in patients with intracranial tumors. Their occurrence and possible pathogenesis. Arch Neurol. 1962. 6: 27-42
6. Glass JP, Jankovic J, Borit A. Hemiballism and metastatic brain tumor. Neurology. 1984. 34: 204-7
7. Hwang KJ, Hong IK, Ahn TB, Yi SH, Lee D, Kim DY. Cortical hemichorea-hemiballism. J Neurol. 2013. 260: 2986-92
8. Iwasa H, Murata Y, Nishimori M, Miyatake K, Tadokoro M, Kohsaki S. Remote effects in the ipsilateral thalamus and/or contralateral cerebellar hemisphere using FDG PET in patients with brain tumors. Jpn J Radiol. 2018. 36: 303-11
9. Krauss JK, Nobbe F, Wakhloo AK, Mohadjer M, Vach W, Mundinger F. Movement disorders in astrocytomas of the basal ganglia and the thalamus. J Neurol Neurosurg Psychiatry. 1992. 55: 1162-7
10. Rana AQ, Yousuf MS, Hashmi MZ, Kachhvi ZM. Hemichorea and dystonia due to frontal lobe meningioma. J Neurosci Rural Pract. 2014. 5: 290-2
11. Salvati M, Frati A, Ferrari P, Verrelli C, Artizzu S, Letizia C. Parkinsonian syndrome in a patient with a pterional meningioma: Case report and review of the literature. Clin Neurol Neurosurg. 2000. 102: 243-5
12. Urasaki E, Tokimura T, Genmoto T, Yokota A. Paroxysmal kinesigenic choreoathetosis associated with frontotemporal arachnoid cyst-case report. Neurol Med Chir (Tokyo). 1999. 39: 169-73
13. Vitek JL, Chockkan V, Zhang JY, Kaneoke Y, Evatt M, DeLong MR. Neuronal activity in the basal ganglia in patients with generalized dystonia and hemiballismus. Ann Neurol. 1999. 46: 22-35
14. Yamada K, Harada M, Inoue N, Yoshida S, Morioka M, Kuratsu J. Concurrent hemichorea and migrainous aura-a perfusion study on the basal ganglia using xenon-computed tomography. Mov Disord. 2008. 23: 425-9