Stereotactic Thalamotomy for Hairdresser's Dystonia: A Case Series

Horisawa, Shiro; Goto, Shinichi; Nakajima, Takeshi; Ochiai, Taku; Kawamata, Takakazu; Taira, Takaomi

doi:10.1159/000446612

Cited by 13 publications

(8 citation statements)

References 27 publications

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“…The Voa contact falls within a Voa-thalamotomy target of 1-5 mm above the AC-PC plane. 16,46 Preoperatively, the right limb tremor was 38 as measured by the Fahn-Tolosa-Marin Tremor Rating Scale. 10 During DBS on and DBS off, tremor was scored as 3 and 41, respectively.…”

Section: Resultsmentioning

confidence: 99%

The effect of unilateral thalamic deep brain stimulation on the vocal dysfunction in a patient with spasmodic dysphonia: interrogating cerebellar and pallidal neural circuits

Poologaindran¹,

Ivanishvili²,

Morrison

et al. 2018

Journal of Neurosurgery

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Spasmodic dysphonia (SD) is a neurological disorder of the voice where a patient's ability to speak is compromised due to involuntary contractions of the intrinsic laryngeal muscles. Since the 1980s, SD has been treated with botulinum toxin A (BTX) injections into the throat. This therapy is limited by the delayed-onset of benefits, wearing-off effects, and repeated injections required every 3 months. In a patient with essential tremor (ET) and coincident SD, the authors set out to quantify the effects of thalamic deep brain stimulation (DBS) on vocal function while investigating the underlying motor thalamic circuitry. A 79-year-old right-handed woman with ET and coincident adductor SD was referred to our neurosurgical team. While primarily treating her limb tremor, the authors studied the effects of unilateral, thalamic DBS on vocal function using the Unified Spasmodic Dysphonia Rating Scale (USDRS) and voice-related quality of life (VRQOL). Since dystonia is increasingly being considered a multinodal network disorder, an anterior trajectory into the left thalamus was deliberately chosen such that the proximal contacts of the electrode were in the ventral oralis anterior (Voa) nucleus (pallidal outflow) and the distal contacts were in the ventral intermediate (Vim) nucleus (cerebellar outflow). In addition to assessing on/off unilateral thalamic Vim stimulation on voice, the authors experimentally assessed low-voltage unilateral Vim, Voa, or multitarget stimulation in a prospective, randomized, doubled-blinded manner. The evaluators were experienced at rating SD and were familiar with the vocal tremor of ET. A Wilcoxon signed-rank test was used to study the pre- and posttreatment effect of DBS on voice. Unilateral left thalamic Vim stimulation (DBS on) significantly improved SD vocal dysfunction compared with no stimulation (DBS off), as measured by the USDRS (p < 0.01) and VRQOL (p < 0.01). In the experimental interrogation, both low-voltage Vim (p < 0.01) and multitarget Vim + Voa (p < 0.01) stimulation were significantly superior to low-voltage Voa stimulation. For the first time, the effects of high-frequency stimulation of different neural circuits in SD have been quantified. Unexpectedly, focused Voa (pallidal outflow) stimulation was inferior to Vim (cerebellar outflow) stimulation despite the classification of SD as a dystonia. While only a single case, scattered reports exist on the positive effects of thalamic DBS on dysphonia. A Phase 1 pilot trial (DEBUSSY; clinical trial no. NCT02558634, clinicaltrials.gov) is underway at the authors' center to evaluate the safety and preliminary efficacy of DBS in SD. The authors hope that this current report stimulates neurosurgeons to investigate this new indication for DBS.

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Section: Resultsmentioning

confidence: 99%

The effect of unilateral thalamic deep brain stimulation on the vocal dysfunction in a patient with spasmodic dysphonia: interrogating cerebellar and pallidal neural circuits

Poologaindran¹,

Ivanishvili²,

Morrison

et al. 2018

Journal of Neurosurgery

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“…Subsequently, several articles have shown the efficacy of vo-thalamotomy for TSFD in musicians, hairdressers, watchmakers, and table tennis players. 7,9,15,19 The vo nucleus of the thalamus is a relay point in the corticobasal ganglia-thalamo-cortical circuit and a major termination site for basal ganglia outputs that control the motor cortex. Circuit abnormalities could induce impaired regulation between desired and unwanted movements, ultimately leading to dystonic movements.…”

Section: Discussionmentioning

confidence: 99%

“…3 TSFD usually appears after years of repetitive and fine movements associated with specific tasks and are seen in musicians, hairdressers, watchmakers, golfers, table tennis players, marksmen, and jugglers. 4–9 TSFD can severely interfere with patients' daily lives, and occupational TSFD can be a risk factor for unemployment if not successfully treated. 2,10…”

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confidence: 99%

“…Although several studies have demonstrated the high efficacy of lesioning on ventro-oral (vo) nucleus of the thalamus (vo-thalamotomy) for TSFDs, these are limited to case reports and a few small-scale studies. 7,9,14–23 We herein retrospectively report on the safety and long-term efficacy of vo-thalamotomy in 171 consecutive patients with TSFD. This is the largest case series involving follow-up study of patients who undergo vo-thalamotomy for TSFD.…”

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Safety and long-term efficacy of ventro-oral thalamotomy for focal hand dystonia

et al. 2019

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ObjectiveTo report the safety and long-term efficacy of ventro-oral thalamotomy for 171 consecutive patients with task-specific focal hand dystonia.MethodsBetween October 2003 and February 2017, 171 consecutive patients with task-specific focal hand dystonia underwent unilateral ventro-oral thalamotomy. Etiologies included writer's cramps (n = 92), musician's dystonias (n = 58), and other occupational task-related dystonias (n = 21). The task-specific focal hand dystonia scale was used to evaluate patients' neurologic conditions (range 1–5, high score indicated a better condition). The scores before surgery; at 1 week, 3 months, and 12 months postoperatively; and the last available follow-up period were determined. Postoperative complications and postoperative recurrence were also evaluated.ResultsThe scores before surgery; at 1 week (1.72 ± 0.57, 4.33 ± 0.85 [p < 0.001]), 3 months (4.30 ± 1.06 [p < 0.001]), and 12 months (4.30 ± 1.13 [p < 0.001]); and the last available follow-up (4.39 ± 1.07 [p < 0.001]) postoperatively improved. The mean clinical follow-up period was 25.4 ± 32.1 months (range: 3–165). Permanent adverse events developed in 6 patients (3.5%). Eighteen patients developed recurrent dystonic symptoms postoperatively. Of these 18 patients, 9 underwent ventro-oral thalamotomy again, of which 7 achieved improvement.ConclusionVentro-oral thalamotomy is a feasible and reasonable treatment for patients with refractory task-specific focal hand dystonias. Prospective, randomized, and blinded studies are warranted to clarify more accurate assessment of the safety and efficacy of ventro-oral thalamotomy for task-specific focal hand dystonia.Classification of evidenceThis study provides Class IV evidence that for patients with task-specific focal hand dystonia, ventro-oral thalamotomy improves dystonia.

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“…Vo thalamotomy has also been offered for musician's and hairdresser dystonia, again another form of TSD. [ 21 22 ] Asahi et al . reported improvement in a patient with table tennis dystonia following Vo thalamotomy.…”

Section: Discussionmentioning

confidence: 99%