The Role of Voice Therapy in the Treatment of Dyspnea and Dysphonia in a Patient With a Vagal Nerve Stimulation Device

The Role of Voice Therapy in the Treatment of Dyspnea and Dysphonia in a Patient With a Vagal Nerve Stimulation Device *Amanda I. Gillespie, *Leah B. Helou, *John W. Ingle, †Maria Baldwin, and *Clark A. Rosen, *yPittsburgh, Pennsylvania Summary: Vagal nerve stimulators (VNS) are implanted to treat medically refractory epilepsy and depression. The VNS stimulates the vagus nerve in the left neck. Laryngeal side effects are common and include dysphagia, dysphonia, and dyspnea. The current case stu
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  The Role of Voice Therapy in the Treatment ofDyspnea and Dysphonia in a Patient With a VagalNerve Stimulation Device *AmandaI.Gillespie,*LeahB.Helou,*JohnW.Ingle,†MariaBaldwin,and*ClarkA.Rosen, * y Pittsburgh, Pennsylvania Summary:  Vagal nerve stimulators (VNS) are implanted to treat medically refractory epilepsy and depression. TheVNS stimulates the vagus nerve in the left neck. Laryngeal side effects are common and include dysphagia, dysphonia,and dyspnea. The current case study represents a patient with severe dyspnea and dysphonia, persisting even with VNSdeactivation. The case demonstrates the use of voice and respiratory retraining therapy for the treatment of VNS-induced dysphonia and dyspnea. It also highlights the importance of a multidisciplinary approach, including laryngol-ogy, neurology, and speech-language pathology, in the treatment of these challenging patients. Key Words:  Respiratory retraining–PVFMD–Vagal nerve stimulation–Epilepsy. INTRODUCTION Vagal nerve stimulation (VNS) devices are implanted in pa-tients with medically refractory epilepsy or in adult patientswithchronicorrecurrentdepression,notrespondingtopharma-cologic antidepressant treatment. The precise physiologicalmechanism of action in decreasing seizure activity and depres-sion is unknown. Implantation of the VNS involves a batteryimplanted on the left chest wall superficial to the pectoral mus-cles and placement of a wire from the stimulator is wrappedaround the left vagus nerve. The device is programmed to cyclethrough ‘‘on’’ and ‘‘off’’ periods of stimulation. Typical on pe-riod is 30 seconds and off period of 5 minutes. Various stimula-tion parameters, such as amplitude of pulse current, stimulationfrequency, and pulse width can also be programed. Typicalstimulation current settings are 1 mA of current with a 30 Hzfrequency and pulse width of 500 microseconds. In addition,the device can be activated to give an extra pulse of stimulationcurrent if a magnet is swiped across the battery. This capabilityallows device stimulation outside the set parameters if the pa-tient or family can appreciate an ‘‘aura’’ or start of a seizure.VNS implantation can have serious adverse effects on voice,swallowing, and breathing. Numerous reports in the literaturedocument the adverse effects of VNS on laryngeal and pharyn-geal function.The most common adverse effect of  VNS is voice distur-bance, occurring in up to 66% of patients. 1 Vocal quality in pa-tients with VNS can be worsened by multiple mechanisms,including vocal fold adduction during stimulation, increasedlaryngopharyngeal contraction during stimulation, and vocalfold paralysis or paresis. Hypertonic pharyngeal and laryngealmuscle contraction during stimulation will not only affect pho-nation but may also alter resonance as well. Varying degrees of airway obstruction can occur with VNS, presenting as dyspnea,stridor, and obstructive sleep apnea. Intermittent partial airwayobstructionmayalsooccursecondarytotransientleftvocalfoldimmobility or hypomobility while the VNS is on, due to con-traction of the left hemilarynx preventing left vocal fold abduc-tion. When the left vocal fold is paralyzed as a sequella of VNSimplantation, patients are at a higher risk for airway obstruc-tion, especially in the event that the right vocal fold becomesimmobile or hypomobile by another etiology (eg, surgery, en-dotracheal intubation).Within the spectrum of partial airway obstruction, paradoxi-cal vocal fold motion (PVFM) is a rare adverse effect of vagalstimulation that is not well described in the literature. PVFMcauses dyspnea due to inappropriate vocal fold adduction dur-inginspiration.WepresentacaseofapatientwithVNSimplan-tation and PVFM-like dyspnea amenable to a combinedapproach of medical and behavioral therapeutic interventions. CASE REPORTLaryngology and speech-language pathologyassessment A 59-year-old male, 4 years post-VNS implantation for medi-cally refractory epileptic seizures, presented with complaintsof dysphonia coinciding with VNS implantation, and dyspneasrcinating approximately 2 years after implantation with noapparent inciting event. Both dysphonia and dyspnea severityincreased substantially with VNS activation. Specifically, thepatient reported an increase in effort with phonation, vocal fa-tigue, and a sensation of ‘‘pulling’’ and ‘‘tightness’’ in the leftside of the neck. The dyspnea required multiple prior hospitaladmissions and was triggered by physical exertion in additionto VNS activation. Flexible laryngoscopy at the initial visitdemonstrated an immobile left vocal fold and a right vocalfold with normal range of motion. When the VNS was turnedon, therewas pronounced contractionof thelefthemilarynx,in-cluding medial contraction of the left false vocal fold. With theVNS on, the glottic opening at rest became narrowed. The rightvocalfold,whichhadnormalrangeofmotionwiththeVNSoff,exhibited hypomobility with decreased abductory range of mo-tion when the VNS was on.At the initial voice clinic visit, the patient completed the Voice Handicap Index-10 (VHI-10) 2 and Dyspnea Index (DI) 3 Accepted for publication August 14, 2013.Financial disclosures: Nothing to disclose.Conflict of interest: None.From the *Department of Otolaryngology - Head and Neck Surgery, University of PittsburghVoiceCenter,UniversityofPittsburghSchoolofMedicine,Pittsburgh,Pennsyl-vania; and the  y Department of Neurology, University of Pittsburgh School of Medicine,Pittsburgh, PennsylvaniaAddresscorrespondenceandreprintrequeststoAmandaI.Gillespie,1400LocustStreet,Bldg B, Suite 11500, Pittsburgh, PA 15219. E-mail: gillespieai@upmc.eduJournal of Voice, Vol. 28, No. 1, pp. 59-610892-1997/$36.00  2014 The Voice Foundation  (Table 1). The VHI-10 measures the level of self-perceived vo-cal handicap, and the DI measures self-perceived dyspnea se-verity. Scores on both measures were elevated compared withnormal controls, indicating high patient-perceived handicapby dysphonia and dyspnea. Following consultation with the pa-tient’s neurologist, the VNS was deactivated due to the severityof his dyspnea and dysphonia. He returned in 3 months withcontinued complaints of dyspnea and dysphonia, indicatinga behavioral component to his symptoms beyond those causedby VNS activation alone. He was also experiencing a return of seizure activity. At this point, the patient was prescribed voiceand respiratory retraining therapy during the ongoing period of VNS deactivation. The purpose of these treatments was to trainrelaxed open laryngeal-pharyngeal postures for breathing anddecrease hyperfunction during phonation. Voice therapy Thepatientunderwentthreeweeklysessionsofvoiceandrespi-ratory retraining therapy. The respiratory retraining was aimedatfacilitatingthemostopenlaryngealairwayandimprovingthepatient’s perceived control over dyspneic episodes. 4,5 Thepatient was trained to exhale for 4 seconds while producingthe voiceless fricative /s/ and then inhale through the mouthwith pursed lips. This manner of breathing was practicedduring rest and with mild exertion (stair climbing, walking).Voice therapy focused on the use of resonant voice. 6 He wastrained to produce forward resonant voice characterized by(1) sensation of anterior facial vibrations and (2) lack of self-perceivedtensioninthethroatoreffortduringphonation.Thesetechniqueswerepracticed attheword,phrase, and,mostimpor-tant, conversational level. Following therapy, the patient wasdischarged from therapy and instructed to return to voice clinicfor re-evaluation. Follow-up evaluations The patient returned for follow-up appointments 3 and 6months after VNS reactivation. VNS settings and patient symp-toms at each follow-up time point are reported in Table 2. Flex-ible laryngoscopy at the second follow-up visit, while the VNSwas in a deactivation mode,revealed that the left vocalfold wasno longer immobile but showed evidence of hypomobility withpurposeful motion. The improvement in left vocal fold range of motion remained stable in the subsequent two follow-up visitswhen the device was off. However, after the VNS was turnedback on at lower settings, a decrease in vocal fold range of mo-tion bilaterally during device activation was observed. Thisfinding indicated that vocal fold range of motion was affectedbilaterally during device activation.At long-term follow-up, 13 months after completion of ther-apy and VNS reactivation, the patient reported ongoing dys-pneic symptoms, which he was able to manage through theuse of respiratory retraining therapy techniques. Use of thesame therapy techniques not only attenuated tonic symptomsof dyspnea but reportedly were preventative to the extent thatthe patient no longer experienced breathing ‘‘attacks.’’ Implications The current case presents the first evidence of contralateraladduction following VNS implantation resulting in dyspneaand dysphonia persisting even throughout a period of VNS de-activation. Three points require further investigation. First,the contralateral adduction observed in this patient could havebeen due to neural cross-innervation via the interarytenoid orGalen’s anastomosis. 7,8 Ipsilateral adduction is expected evenat the lowest pulse duration levels (0.025–0.50 mA) butcontralateral vocal fold adduction is not anticipated at suchlow levels. 9 However, in the present case, the VNS was acti-vated at large amplitudes (0.75 mA), which theoretically mightinduce contralateral adduction due to bidirectional signal trans-port not only directly to the left recurrent laryngeal nerve butalso from efferent brainstem reflexes as a result of stimulation.VNS stimulation of 10 Hz (the patient’s frequency setting) isnotexpectedtotriggeradduction,especiallyofthecontralateralvocal fold. 10 TABLE 2.VNS Setting and Subjective Patient Complaint at Each Time Point Visit # Time VNS Settings Symptoms1 Initial clinic visit Output: 1.0 mAFrequency: 20 HzOn: 30 s; off: 3 minSevere dysphonia and dyspnea. Bothworsened with device ‘‘on’’2 3 mo posttherapy1 wk post-VNS reactivationOutput: 0.25 mAFrequency: 10 HzOn: 14 s; off: 5 minDysphoniaanddyspneawithVNSstimulationand with exertion (absent stimulation)3 6 mo posttherapy3 mo post-VNS reactivationOutput: 0.75 mAFrequency: 10 HzOn: 14 s; off : 5 minDyspnea and dysphonia were ‘‘90% better’’Telephone follow-up 13 mo posttherapy10 mo post-VNS reactivationOutput: 0.75 mAFrequency: 10 HzOn: 14 s; off : 5 minAwareness of symptoms but able to usetherapy techniques to ‘‘manage’’ and‘‘avoid attacks’’ TABLE 1.VHI-10 and DI Scores for Each Time Point Visit # VHI-10 (Max: 40) DI (Max: 40)1 33 332 (3 mo post-tx) 28 263 (6 mo post-tx) 24 23Journal of Voice, Vol. 28, No. 1, 2014 60  A second point of interest is that the persistence of the pa-tient’s symptoms even after VNS deactivation may provide in-sight to mechanisms of PVFM in patients following noxiousexposure. Some have hypothesized that central neuronalchanges occur with repeated noxious stimuli to the larynx, re-sulting in PVFM even after cessation of the stimuli. 11 In thiscase, the cyclic VNS activation triggering vocal fold adductionmayhavecausedneurologicchangesatthecentralorperipherallevel resulting in ongoing laryngeal hyperadduction causingdyspnea even after VNS deactivation.A third point of interest relates to respiratory retraining asa known effective treatment for dyspnea due to PVFM. 5,12–14 Behavioral treatment of dyspnea caused by inappropriatevocal fold adduction in relation to VNS activation has notpreviously been reported in the literature. Typical treatmentsfor VNS-induced dyspnea involve device titration or deactiva-tion. 15 This case demonstrates the usefulness of behavioral in-tervention in overriding VNS-induced dyspnea.In conclusion, respiratory retraining therapy was successfulin alleviating symptoms of PVFM-like dyspnea in a patientwith VNS. Multidisciplinary teamwork of laryngology, neurol-ogy, and speech-language pathology is critical in the manage-ment of concomitant dyspnea and epilepsy. REFERENCES 1. Handforth A, DeGiorgio CM, Schachter SC, et al. Vagus nerve stimulationtherapyforpartial-onsetseizures:arandomizedactive-controltrial.  Neurol-ogy . 1998;51:48–55.2. Rosen CA, Lee AS, Osborne J, Zullo T, Murry T. Development and valida-tion of the voice handicap index-10.  Laryngoscope . 2004;114:1549–1556.3. Shembel A, Gartner-Schmidt J, Rosen CA, Zullo TG. Two novel instru-ments: development and validation of the Dyspnea Index (DI) and CoughSeverity Index (CSI). The Voice Foundation Annual Symposium; June 5,2011; 2011; Philadelphia, PA.4. Blager FB, Gay ML, Wood RP. Voice therapy techniques adapted to treat-ment of habit cough: a pilot study.  J Commun Disord  . 1988;21:393–400.5. Murry T, Tabaee A, Owczarzak V, Aviv JE. Respiratory retraining therapyand management of laryngopharyngeal reflux in the treatment of patientswithcoughandparadoxicalvocalfoldmovementdisorder.  AnnOtolRhinol Laryngol . 2006;115:754–758.6. Verdolini K, Druker DG, Palmer PM, Samawi H. Laryngeal adduction inresonant voice.  J Voice . 1998;12:315–327.7. Martin-Oviedo C, Maranillo E, Lowy-Benoliel A, et al. Functional role of human laryngeal nerve connections.  Laryngoscope . 2011;121:2338–2343.8. Sanudo JR, Maranillo E, Leon X, Mirapeix RM, Orus C, Quer M. An ana-tomical study of anastomosesbetween the laryngeal nerves.  Laryngoscope .1999;109:983–987.9. Ardesch JJ, Sikken JR, Veltink PH, van der Aa HE, Hageman G,Buschman HP. Vagus nerve stimulation for epilepsy activates the vocalfolds maximally at therapeutic levels.  Epilepsy Res . 2010;89:227–231.10. LundyDS,CasianoRR,LandyHJ,GalloJ,GalloB,RamseyRE.Effectsof vagal nerve stimulation on laryngeal function.  J Voice . 1993;7:359–364.11. Morrison M, Rammage L, Emami AJ. The irritable larynx syndrome.  J Voice . 1999;13:447–455.12. Hatzelis V, Murry T. Paradoxical vocal fold motion: respiratory retrainingto manage long-term symptoms.  J Soc Bras Fonoaudiol . 2012;24:80–85.13. Murry T, Sapienza C. The role of voice therapy in the management of par-adoxical vocal fold motion, chroniccough, and laryngospasm.  OtolaryngolClin North Am . 2010;43:73–83 [viii-ix].14. Rameau A, Foltz RS, Wagner K, Zur KB. Multidisciplinary approach tovocal cord dysfunction diagnosis and treatment in one session: a singleinstitutional outcome study.  Int J Pediatr Otorhinolaryngol . 2012;76:31–35.15. Bhatt YM, Hans PS, Belloso A. Airway compromise secondary to vagusnerve stimulator: case report and implications for otolaryngologists.  J Lar- yngol Otol . 2010;124:557–559. Amanda I. Gillespie,  et al   Respiratory Retraining for VNS-Induced PVFMD  61
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