Deep Brain Stimulation for Treatment of Voice Disorders Mary J. Hawkshaw and Robert T. Sataloff, Philadelphia, Pennsylvania Summary: Objectives. Vocal tremor is a common, troublesome disorder that is difficult to treat. Efficacy of deep brain stimulation (DBS) was reported more than a decade ago. Most laryngologists are not familiar with the technique or its potential. This review was undertaken to assemble relevant literature written over the past decade and assess the clinical implications of that literature. Design. Literature review. Methods. PubMed search from 2002 through 2011. Results. A small number of articles on the topic have been identified, some of which appear to provide information of potential clinical importance for voice patients. Conclusion. A review of the literature from 2002 through 2011 has revealed several studies supporting the efficacy of DBS as well as adverse consequences of specific technical approaches (such as high-frequency DBS). In the aging population, the prevalence of this voice tremor is likely to increase. We suspect that DBS may be underused; and laryngologists should collaborate with neurosurgeons, speech-language pathologists, and voice scientists to study more extensively the safety and efficacy of DBS for treatment of voice disorders. Key Words: Deep brain stimulation–Dysphonia–Vocal fold tremor.
In 2002, we published our experience with deep brain stimulation (DBS) in the treatment of two patients with vocal tremor.1 Our results not only suggested that DBS may be helpful in controlling vocal tremor but also that more research was needed. In 2002, when we examined the literature, we found that very little had been published on the use of DBS on vocal tremor. The purpose of this review was to evaluate the published data since 2002 on DBS in the treatment of voice and potentially related disorders. A PubMed literature search was done using the keywords: DBS, voice disorders, dystonias, laryngeal dystonia, thalamic DBS, voice tremor, and spasmodic dysphonia. Thalamic stimulation for the reduction of tremor was first reported in 1991 by Benabid et al.2 Since that time, many reports have been published on the successful use of DBS for neurological movement disorders, including Parkinson’s disease, essential tremor, and dystonias.3–24 In addition to treating severe motor disorders, DBS has been used in the management of chronic pain, and recent studies suggest its application potential for treating psychiatric illnesses, including obsessivecompulsive disorder and depression.3,4 DBS has proven to be effective in the management of patients with hyperkinetic movement disorders, such as Gilles de la Tourette’s syndrome and tardive dyskinesia, tics, and in cervical dystonia refractory to medical treatment.5–9,24 In 2010, Thevathasan and Gregory25 reported that DBS is now used routinely as a treatment option for patients with advanced Parkinson’s disease, primary dystonias, secondary dystonias, and essential tremor. Accepted for publication May 15, 2012. From the Department of Otolaryngology—Head and Neck Surgery, Drexel University College of Medicine, Philadelphia, Pennsylvania. Address correspondence and reprint requests to Robert T. Sataloff, Department of Otolaryngology—Head and Neck Surgery, Drexel University College of Medicine, 1721 Pine Street, Philadelphia, PA 19103. E-mail:
[email protected] Journal of Voice, Vol. 26, No. 6, pp. 769-771 0892-1997/$36.00 Ó 2012 The Voice Foundation doi:10.1016/j.jvoice.2012.05.004
Focal dystonias are specific to the body part affected: blepharospasm (eyes), writer’s cramp (hand), cervical (neck), spasmodic dysphonia (larynx), and oromandibular dystonia (lower face and mouth). The most common task-specific dystonias are laryngeal and writer’s cramp. DBS has been used and proven effective in the treatment of medically intractable focal dystonias. Cho et al26 found DBS effective in the treatment of writer’s cramp. Researchers in Korea reported their experience in treating 11 patients with Meige’s syndrome refractory to medical management.27 They reported postsurgical improvement in oromandibular dystonia, blepharospasm, and speech and swallowing. In 2010, Romito et al28 reported their experience in treating one patient with severe Meige’s syndrome with cervical brachial involvement. This patient underwent a bilateral ventroposterolateral globus pallidus internus implant followed by low voltage brain stimulation. Follow-up in 38 months showed sustained improvement and control of the dystonia without changing the electrical settings of the implants postoperatively. Researchers at the University of Kansas Medical Center reported their findings on the effect of DBS of the ventral intermediate nucleus (VIM) of the thalamus on the voice tremor in seven patients implanted primarily for management of hand tremor.29 They reported reduction of the voice tremor in four of the seven patients. However, they also reported that the gains in voice improvement did not parallel improvements of the upper extremities. In 1999, Taha et al30 evaluated their results using bilateral thalamic DBS in treating head, voice, and bilateral limb tremor associated with Parkinson’s disease (six patients), essential tremor (15 patients), and multiple sclerosis (two patients). Ninety percent of the patients with severe head tremor and 86% of the patients with voice tremor showed improvement within a mean follow-up of 10 months. These researchers suggested that head and voice tremors are the primary indications for this surgical procedure.
770 Moringlane et al31 reported the case of an 81-year-old female disabled by head, voice, and upper extremity tremor. She was treated with bilateral thalamic, intermediate nucleus (VIM) stimulation. With the stimulator on, they achieved complete normalization of her voice and improved vocal fold vibration. Their acoustic and electroglottographic analysis revealed hyperfunctional voicing with the stimulator both on and off. In 2010, researchers in the Netherlands reported their findings in a group of patients with DYT6 dystonia and their response to DBS. Four patients had segmental dystonia, oromandibular and laryngeal involvement. With DBS, these patients achieved moderate-to-good improvement in motor function and only marginal improvement in speech.32 Parkinson’s disease is a progressively debilitating neurological disease that results in impaired motor control of extremities and deficits of voice, speech, and swallowing. Earlier, DBS management of Parkinson’s disease has included medications to treat the symptoms and speech therapy. In 2000, researchers at the University of Florida/Gainesville33 reviewed the literature and examined the treatment options for patients with Parkinson’s disease and the effects on speech. They found that combined treatment with medications and speech therapy improved both the speaking voice and speech. They suggested that pallidotomy and DBS might be effective in the management of Parkinson’s but that further research and experience was needed. They also stressed that efforts should focus on combined therapy approaches. Over the past decade, other researchers have studied the effects of electrostimulation of the subthalamic nucleus (STN) on the speech subsystems and on the respiratory/phonatory subsystems in patients with Parkinson’s disease.34–37 D’Alatri et al38 studied the effect of bilateral STN stimulation and medication on Parkinson’s disease and dysarthria in 12 patients (11 male and one female). Neurological assessments and acoustic recordings of the voice were made. They found that STN improved motor function and voice tremor noting a ‘‘major stability to glottal vibration.’’ Similar to other reports, they noted that STN had a greater effect on the motor dysfunction of extremities than on the voice dysfunction. However, they reported no adverse effect on speech intelligibility. In 2010, Hammer et al39 published their research findings on the changes in speech, respiratory, and laryngeal control in 18 patients with Parkinson’s disease after STN-DBS. They used aerodynamic measures of speech and respiration to assess changes after DBS. Their findings showed that high-frequency STN-DBS often caused ‘‘respiratory overdrive and excessive vocal fold closure’’ and thus might be less helpful in controlling speech and laryngeal symptoms. Their article also provides an extensive list of references that highlights the number of other published reports on the effects of DBS on speech and voice. In 2008, Zealear’s group proposed another potential use for DBS. They studied the effects of DBS in canines with vocal fold paralysis. They assessed the effects in animals with complete denervation and those with synkinetic reinnervation and found DBS to be effective at reanimating the posterior cricoarytenoid muscle in both conditions.40
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DBS is a neurosurgical procedure and, like all others, is associated with risks as well as benefits. Hardware complications, venous air emboli, intracranial bleeding, infection, electrode malposition, and erosion have been reported.41–43 Blomstedt et al44 reported moderate dysarthria and severe dysphonia in three of five patients after DBS. Tripoliti et al45 noted decreased speech intelligibility after high-voltage DBS that resulted in improved motor function. They cautioned that strict parameters are needed in speech motor control. Interestingly, acquired stuttering induced by thalamic DBS also has been reported.46,47 Literature over the last decade has supported the efficacy of DBS for treatment of vocal tremor. However, there have been few studies, and the number of patients treated remains small. Voice tremor can be a troublesome symptom; and it sometimes can be disabling. Medical treatment and voice therapy are often not sufficient to control symptoms especially in patients with severe tremor. The good results reported in some cases suggest that this treatment modality may be underused. Patients have an intuitive reluctance to undergo ‘‘brain surgery.’’ However, DBS is a common, reasonably safe procedure; and additional research should be encouraged to determine its safety and efficacy in treatment of patients with voice tremor. REFERENCES 1. Sataloff RT, Heuer RJ, Munz M, Yoon MS, Spiegel JR. Vocal tremor reduction with deep brain stimulation: a preliminary report. J Voice. 2002;16: 132–135. 2. Benabid AL, Pollak P, Dongming G, et al. Chronic electrical stimulation of the ventralis intermedius nucleus of the thalamus as a treatment of movement disorders. J Neurosurg. 1996;84:203–214. 3. Dowling J. Deep brain stimulation: current and emerging indications. Mo Med. 2008;105:424–428. 4. Clausen J. Ethical brain stimulation—neuroethics of deep brain stimulation in research and clinical practice. Eur J Neurosci. 2010;32:1152–1162. 5. Welter ML, Grabli D, Vidailhet M. Deep brain stimulation for hyperkinetics disorders: dystonia, tardive dyskinesia, and tics. Curr Opin Neurol. 2010;23:420–425. 6. Gruber D, Trottenberg T, Kivi A, et al. Long-term effects of pallidal deep brain stimulation in tardive dystonia. Neurology. 2009;73:53–58. 7. Sako W, Goto S, Shimazu H, et al. Bilateral deep brain stimulation of the globus pallidus internus in tardive dystonia. Mov Disord. 2008;23:1929–1931. 8. Kefalopoulou Z, Paschali A, Markaki E, Vassilakos P, Ellul J, Constantoyannis C. A double-blind study on a patient with tardive dyskinesis treated with pallidal deep brain stimulation. Acta Neurol Scand. 2009; 119:269–273. 9. Cacciola F, Farah JO, Eldridge PR, Byrne P, Varma TK. Bilateral deep brain stimulation for cervical dystonia: long-term outcome in a series of 10 patients. Neurosurgery. 2010;67:957–963. 10. Torres CV, Moro E, Dostrovsky JO, Hutchinson WD, Poon YY, Hodale M. Unilateral pallidal deep brain stimulation in a patient with cervical dystonia and tremor. J Neurosurg. 2010;113:1230–1233. 11. Krauss JK. Surgical treatment of dystonia. Eur J Neurol. 2010;17(suppl 1): 97–101. 12. Oropilla JQ, Diesta CC, Itthimathin P, Suchowesky O, Kiss ZH. Both thalamic and pallidal deep brain stimulation for myoclonic dystonia. J Neurosurg. 2010;112:1267–1270. 13. Albanese A, Asmus F, Bhatia KP, et al. EFNS guidelines on diagnosis and treatment of primary dystonias. Eur J Neurol. 2011;18:5–18. 14. Valldeoriola F, Regidor I, Minguez-Castellanos A, et al. Efficacy and safety of pallidal stimulation in primary dystonia: results of the Spanish multicentric study. J Neurol Neurosurg Psychiatry. 2010;81:65–69. 15. Shanker V, Bressman SB. What’s new in dystonia? Curr Neurol Neurosci Rep. 2009;9:278–284.
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