- Department of Neurosurgery, Tokyo Women’s Medical University, Tokyo, Japan.
Shiro Horisawa, Department of Neurosurgery, Tokyo Women’s Medical University, Tokyo, Japan.
DOI:10.25259/SNI_840_2022Copyright: © 2022 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, transform, 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: Shiro Horisawa, Takakazu Kawamata, Takaomi Taira. Seven-year resolution of cervical dystonia after unilateral pallidotomy: A case report. 23-Dec-2022;13:586
How to cite this URL: Shiro Horisawa, Takakazu Kawamata, Takaomi Taira. Seven-year resolution of cervical dystonia after unilateral pallidotomy: A case report. 23-Dec-2022;13:586. Available from: https://surgicalneurologyint.com/?post_type=surgicalint_articles&p=12079
Background: Reports on the long-term effects of pallidotomy for cervical dystonia remain scarce.
Case Description: We report a case of cervical dystonia successfully treated by unilateral pallidotomy. The patient was a 29-year-old man without past medical and family history of cervical dystonia. At the age of 28 years, neck rotation to the right with right shoulder elevation developed and gradually became worse. After symptoms failed to respond to repetitive botulinum toxin injections and oral medications, he underwent left pallidotomy, which resulted in significant improvement of cervical dystonia and shoulder elevation without surgical complications. At the 3-month evaluation, the symptoms completely improved. The Toronto Western Spasmodic Torticollis Rating Scale score dramatically improved from 39 points before surgery to 0 points at 7-year postoperative evaluation.
Conclusion: This case suggests that unilateral pallidotomy can be an alternative treatment option for cervical dystonia.
Keywords: Cervical dystonia, Globus pallidus internus, Pallidotomy
Cervical dystonia is an involuntary movement disorder, in which involuntary muscle contractions in the cervical musculature cause postural abnormalities.[
A 29-year-old man was diagnosed with cervical dystonia at the age of 27 years after developing symptoms of cervical rotation to the right and consulted a neurologist. He received medications (baclofen 15 mg, trihexyphenidyl 6 mg), but his symptoms were not improved. In addition, he was treated several times with botulinum toxin injections with no response. He was referred to our hospital for surgical treatment. The patient had right cervical rotation of 45–60°, associated neck pain, and elevation of the right shoulder: dystonic symptoms were alleviated by holding the posterior neck with the hand (sensory tricks). On awakening, dystonia symptoms were relatively mild, but gradually worsened from noon to evening (early morning effect). No other movement disorders were observed. Based on the characteristic findings of dystonia, such as sensory tricks and early morning effect, cervical dystonia was diagnosed [
As the patient’s cervical dystonia symptoms were accompanied by symptoms of the right shoulder elevation, selective peripheral denervation was not a treatment option. The patient strongly rejected implantation of the instrument and preferred to undergo RF surgery. As the affected area of dystonia was the right shoulder and right cervical rotation, we decided to perform left pallidotomy based on the symptomatic predominance of involuntary motor output from the left hemisphere. Under local anesthesia, a head magnetic resonance imaging (MRI; T1-axial/T2-coronal image) examination was performed with a Leksell frame, and a surgical plan was made using a Leksell surgiplan (Elekta AB; Stockholm, Sweden). The left GPi was set 19.5 mm to the left lateral, 3.0 mm inferior, and 2.0 mm anterior to the midpoint of the anterior commissure and posterior commissure. A coagulation probe with a heat-conducting part of 1 mm in diameter and 4 mm in length was used, and a Leksell Neurogenerator (Elekta AB) was used for electrical stimulation and thermal coagulation. Before thermal coagulation, electrical stimulation (130 µs/100 Hz/2–5 mA) was performed to confirm that there was no proximity to the optic tract or internal capsule. Thermal coagulation was performed at 70°C for 40 s. Then, the electrode was withdrawn in 3-mm increments to increase the lesion size, producing two contiguous lesions. Electrical stimulation and thermal coagulation were also performed 3 mm postero-laterally from the target. Therefore, in total, four lesions were made using two trajectories. The volume of the lesions was measured using a 3D slicer on a T1-weighted MRI performed immediately postoperatively. The measured volume was 99.3 mm3. The first trajectory lesion was confirmed at 19.7 mm lateral, 2.3 mm anterior, and 3.0 mm inferior to the midpoint of AC-PC. The second trajectory lesion was confirmed at 20.5 mm lateral, 0.3 mm posterior, and 2.0 mm inferior to the midpoint of AC-PC.
From the day after surgery, the neck was maintained in the midline position, and right shoulder elevation was improved to the point where there was no difference between the left and right sides [
In this case, unilateral pallidotomy for cervical dystonia resulted in complete symptomatic improvement over 7 years. This is the report with the longest follow-up period of pallidotomy for cervical dystonia. The current standard surgical treatment is bilateral GPi-DBS, but before the advent of DBS, thermal coagulation surgery using various targets was used in the 1960s–1980s.[
GPi-DBS for cervical dystonia is generally performed bilaterally. Cervical dystonia is often accompanied by symptoms of the left/right rotation/tilting, and involuntary muscle contractions in the cervical muscle groups are often not symmetrical. The condition may also be associated with involuntary movements, such as shoulder elevation, upper extremity dystonia, or tremor, and one of the left or right cerebral hemispheres is often presumed to be the more symptomatically dominant hemisphere. Indeed, interhemispheric differences in neural activity in GPi have been reported to be present in rotational cervical dystonia but absent in symmetrical retroflex cervical dystonia. Abnormalities in cortical excitability and inhibition with transcranial magnetic stimulation have been identified in dystonia cases.[
In the case of such asymmetrical rotator cervical dystonia, there has been no debate concerning whether treatment should be directed to the bilateral cerebral hemispheres. The effects of unilateral GPi-DBS for rotatory cervical dystonia are scattered in case reports.[
Complications of pallidotomy are known to increase in frequency and severity, especially when performed bilaterally.[
This case suggests that unilateral pallidotomy can be an alternative treatment option for cervical dystonia.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent.
Financial support and sponsorship
This work was supported by JSPS KAKENHI Grant Number JP21K09113.
Conflicts of interest
There are no conflicts of interest.
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Despite a long history of destructive interventions in the surgical treatment of movement disorders, it remains unclear if the disease progression, natural healing and brain plasticity translate into gradual worsening of surgical outcomes with longer duration of follow up. Here the authors of the report present a complete resolution of cervical dystonia in a patient treated with radiofrequency thermoablation of conventional pallidal target with torticollis rating (TWSTRS) decreasing from 39 before surgery to 4 immediately after surgery and 0 at the time of 7-year follow up. The correct matching of clinical indications with defined surgical target and appropriate patient and procedure selection are likely the reasons for such positive outcome. This result supports validity of considering targeted lesioning along with an option of deep brain stimulation in carefully selected patients and in presence of an experienced surgical team.
Konstantin Slavin, MD
The views and opinions expressed in this article are those of the authors and do not necessarily reflect the official policy or position of the Journal or its management. The information contained in this article should not be considered to be medical advice; patients should consult their own physicians for advice as to their specific medical needs.
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