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Deep Brain Stimulation for Treatment of Voice Disorders

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Page 1: Deep Brain Stimulation for Treatment of Voice Disorders

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

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laryngoloStreet, PhJourna0892-1� 201doi:10

brain stimulation (DBS) was reported more than a decade ago. Most laryngologists are not familiar with the techniqueor its potential. This review was undertaken to assemble relevant literature written over the past decade and assess theclinical 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 ofpotential clinical importance for voice patients.Conclusion. A review of the literature from 2002 through 2011 has revealed several studies supporting the efficacy ofDBS as well as adverse consequences of specific technical approaches (such as high-frequency DBS). In the aging pop-ulation, the prevalence of this voice tremor is likely to increase. We suspect that DBS may be underused; and laryngol-ogists should collaborate with neurosurgeons, speech-language pathologists, and voice scientists to study moreextensively 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 stimula-tion (DBS) in the treatment of two patients with vocal tremor.1

Our results not only suggested that DBS may be helpful in con-trolling vocal tremor but also that more research was needed. In2002, when we examined the literature, we found that very littlehad been published on the use of DBS on vocal tremor.

The purpose of this review was to evaluate the published datasince 2002 on DBS in the treatment of voice and potentially re-lated disorders. A PubMed literature search was done using thekeywords: DBS, voice disorders, dystonias, laryngeal dystonia,thalamic DBS, voice tremor, and spasmodic dysphonia.

Thalamic stimulation for the reduction of tremor was first re-ported in 1991 by Benabid et al.2 Since that time, many reportshave been published on the successful use of DBS for neurolog-ical movement disorders, including Parkinson’s disease, essen-tial tremor, and dystonias.3–24 In addition to treating severemotor disorders, DBS has been used in the management ofchronic pain, and recent studies suggest its applicationpotential for treating psychiatric illnesses, including obsessive-compulsive disorder and depression.3,4

DBS has proven to be effective in the management of patientswith hyperkinetic movement disorders, such as Gilles de laTourette’s syndrome and tardive dyskinesia, tics, and in cervicaldystonia refractory to medical treatment.5–9,24

In 2010, Thevathasan and Gregory25 reported that DBS isnow used routinely as a treatment option for patients with ad-vanced Parkinson’s disease, primary dystonias, secondary dys-tonias, and essential tremor.

ted for publication May 15, 2012.he Department of Otolaryngology—Head and Neck Surgery, Drexel Universityf Medicine, Philadelphia, Pennsylvania.ss correspondence and reprint requests to Robert T. Sataloff, Department of Oto-gy—Head and Neck Surgery, Drexel University College of Medicine, 1721 Pineiladelphia, PA 19103. E-mail: [email protected] of Voice, Vol. 26, No. 6, pp. 769-771997/$36.002 The Voice Foundation.1016/j.jvoice.2012.05.004

Focal dystonias are specific to the body part affected: bleph-arospasm (eyes), writer’s cramp (hand), cervical (neck), spas-modic dysphonia (larynx), and oromandibular dystonia (lowerface and mouth). The most common task-specific dystoniasare laryngeal and writer’s cramp. DBS has been used andproven effective in the treatment of medically intractable focaldystonias. Cho et al26 found DBS effective in the treatment ofwriter’s cramp. Researchers in Korea reported their experiencein treating 11 patients with Meige’s syndrome refractory tomedical management.27 They reported postsurgical improve-ment in oromandibular dystonia, blepharospasm, and speechand swallowing.

In 2010, Romito et al28 reported their experience in treatingone patient with severe Meige’s syndrome with cervical bra-chial involvement. This patient underwent a bilateral ventro-posterolateral globus pallidus internus implant followed bylow voltage brain stimulation. Follow-up in 38 months showedsustained improvement and control of the dystonia withoutchanging the electrical settings of the implants postoperatively.

Researchers at the University of Kansas Medical Center re-ported their findings on the effect of DBS of the ventral interme-diate nucleus (VIM) of the thalamus on the voice tremor inseven patients implanted primarily for management of handtremor.29 They reported reduction of the voice tremor in fourof the seven patients. However, they also reported that the gainsin voice improvement did not parallel improvements of the up-per extremities.

In 1999, Taha et al30 evaluated their results using bilateralthalamic DBS in treating head, voice, and bilateral limb tremorassociated with Parkinson’s disease (six patients), essentialtremor (15 patients), and multiple sclerosis (two patients).Ninety percent of the patients with severe head tremor and86% of the patients with voice tremor showed improvementwithin a mean follow-up of 10 months. These researchers sug-gested that head and voice tremors are the primary indicationsfor this surgical procedure.

Page 2: Deep Brain Stimulation for Treatment of Voice Disorders

Journal of Voice, Vol. 26, No. 6, 2012770

Moringlane et al31 reported the case of an 81-year-old femaledisabled by head, voice, and upper extremity tremor. She wastreated with bilateral thalamic, intermediate nucleus (VIM)stimulation. With the stimulator on, they achieved completenormalization of her voice and improved vocal fold vibration.Their acoustic and electroglottographic analysis revealedhyperfunctional voicing with the stimulator both on and off.

In 2010, researchers in the Netherlands reported their find-ings in a group of patients with DYT6 dystonia and theirresponse to DBS. Four patients had segmental dystonia, oro-mandibular and laryngeal involvement. With DBS, thesepatients achieved moderate-to-good improvement in motorfunction and only marginal improvement in speech.32

Parkinson’s disease is a progressively debilitating neurolog-ical disease that results in impaired motor control of extremitiesand deficits of voice, speech, and swallowing. Earlier, DBSmanagement of Parkinson’s disease has included medicationsto treat the symptoms and speech therapy. In 2000, researchersat the University of Florida/Gainesville33 reviewed the litera-ture and examined the treatment options for patients with Par-kinson’s disease and the effects on speech. They found thatcombined treatment with medications and speech therapy im-proved both the speaking voice and speech. They suggestedthat pallidotomy and DBS might be effective in the manage-ment of Parkinson’s but that further research and experiencewas needed. They also stressed that efforts should focus oncombined therapy approaches.

Over the past decade, other researchers have studied the ef-fects of electrostimulation of the subthalamic nucleus (STN)on the speech subsystems and on the respiratory/phonatory sub-systems in patients with Parkinson’s disease.34–37

D’Alatri et al38 studied the effect of bilateral STN stimula-tion and medication on Parkinson’s disease and dysarthria in12 patients (11male and one female). Neurological assessmentsand acoustic recordings of the voice were made. They foundthat STN improved motor function and voice tremor notinga ‘‘major stability to glottal vibration.’’ Similar to other reports,they noted that STN had a greater effect on the motor dysfunc-tion of extremities than on the voice dysfunction. However, theyreported no adverse effect on speech intelligibility.

In 2010, Hammer et al39 published their research findings onthe changes in speech, respiratory, and laryngeal control in 18patients with Parkinson’s disease after STN-DBS. They usedaerodynamic measures of speech and respiration to assesschanges after DBS. Their findings showed that high-frequencySTN-DBS often caused ‘‘respiratory overdrive and excessivevocal fold closure’’ and thus might be less helpful in controllingspeech and laryngeal symptoms. Their article also providesan extensive list of references that highlights the number ofother published reports on the effects of DBS on speech andvoice.

In 2008, Zealear’s group proposed another potential use forDBS. They studied the effects of DBS in canines with vocalfold paralysis. They assessed the effects in animals with com-plete denervation and those with synkinetic reinnervation andfound DBS to be effective at reanimating the posterior cricoar-ytenoid muscle in both conditions.40

DBS is a neurosurgical procedure and, like all others, is asso-ciated with risks as well as benefits. Hardware complications,venous air emboli, intracranial bleeding, infection, electrodemalposition, and erosion have been reported.41–43 Blomstedtet al44 reported moderate dysarthria and severe dysphonia inthree of five patients after DBS. Tripoliti et al45 noted decreasedspeech intelligibility after high-voltage DBS that resulted in im-proved motor function. They cautioned that strict parametersare needed in speech motor control. Interestingly, acquired stut-tering induced by thalamic DBS also has been reported.46,47

Literature over the last decade has supported the efficacy ofDBS for treatment of vocal tremor. However, there have beenfew studies, and the number of patients treated remains small.Voice tremor can be a troublesome symptom; and it sometimescan be disabling. Medical treatment and voice therapy are oftennot sufficient to control symptoms especially in patients with se-vere tremor. The good results reported in some cases suggestthat this treatment modality may be underused. Patients havean intuitive reluctance to undergo ‘‘brain surgery.’’ However,DBS is a common, reasonably safe procedure; and additional re-search should be encouraged to determine its safety and efficacyin treatment of patients with voice tremor.

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