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 study represents a patient with severe dyspnea and dysphonia, persisting even with VNS
deactivation. 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 retrainingPVFMDVagal nerve stimulationEpilepsy.
INTRODUCTION
Vagal nerve stimulation (VNS) devices are implanted in pa-
tients with medically refractory epilepsy or in adult patients
with chronic or recurrent depression, not responding to pharma-
cologic antidepressant treatment. The precise physiological
mechanism of action in decreasing seizure activity and depres-
sion is unknown. Implantation of the VNS involves a battery
implanted on the left chest wall supercial to the pectoral mus-
cles and placement of a wire from the stimulator is wrapped
around the left vagus nerve. The device is programmed to cycle
through 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, stimulation
frequency, and pulse width can also be programed. Typical
stimulation current settings are 1 mA of current with a 30 Hz
frequency and pulse width of 500 microseconds. In addition,
the device can be activated to give an extra pulse of stimulation
current if a magnet is swiped across the battery. This capability
allows 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 literature
document 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, increased
laryngopharyngeal contraction during stimulation, and vocal
fold paralysis or paresis. Hypertonic pharyngeal and laryngeal
muscle 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 airway
obstruction may also occur secondary to transient left vocal fold
immobility 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 VNS
implantation, patients are at a higher risk for airway obstruc-
tion, especially in the event that the right vocal fold becomes
immobile 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 vagal
stimulation that is not well described in the literature. PVFM
causes dyspnea due to inappropriate vocal fold adduction dur-
ing inspiration. We present a case of a patient with VNS implan-
tation and PVFM-like dyspnea amenable to a combined
approach of medical and behavioral therapeutic interventions.
CASE REPORT
Laryngology and speech-language pathology
assessment
A 59-year-old male, 4 years post-VNS implantation for medi-
cally refractory epileptic seizures, presented with complaints
of dysphonia coinciding with VNS implantation, and dyspnea
originating approximately 2 years after implantation with no
apparent inciting event. Both dysphonia and dyspnea severity
increased substantially with VNS activation. Specically, the
patient reported an increase in effort with phonation, vocal fa-
tigue, and a sensation of pulling and tightness in the left
side of the neck. The dyspnea required multiple prior hospital
admissions and was triggered by physical exertion in addition
to VNS activation. Flexible laryngoscopy at the initial visit
demonstrated an immobile left vocal fold and a right vocal
fold with normal range of motion. When the VNS was turned
on, there was pronounced contraction of the left hemilarynx, in-
cluding medial contraction of the left false vocal fold. With the
VNS on, the glottic opening at rest became narrowed. The right
vocal fold, which had normal range of motion with the VNS off,
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.
Conict of interest: None.
From the *Department of Otolaryngology - Head and Neck Surgery, University of
Pittsburgh Voice Center, University of Pittsburgh School of Medicine, Pittsburgh, Pennsyl-
vania; and the yDepartment of Neurology, University of Pittsburgh School of Medicine,
Pittsburgh, Pennsylvania
Address correspondence and reprint requests to Amanda I. Gillespie, 1400 Locust Street,
Bldg B, Suite 11500, Pittsburgh, PA 15219. E-mail: gillespieai@upmc.edu
Journal of Voice, Vol. 28, No. 1, pp. 59-61
0892-1997/$36.00
2014 The Voice Foundation
http://dx.doi.org/10.1016/j.jvoice.2013.08.007
(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 with
normal controls, indicating high patient-perceived handicap
by dysphonia and dyspnea. Following consultation with the pa-
tients neurologist, the VNS was deactivated due to the severity
of his dyspnea and dysphonia. He returned in 3 months with
continued complaints of dyspnea and dysphonia, indicating
a behavioral component to his symptoms beyond those caused
by VNS activation alone. He was also experiencing a return of
seizure activity. At this point, the patient was prescribed voice
and respiratory retraining therapy during the ongoing period of
VNS deactivation. The purpose of these treatments was to train
relaxed open laryngeal-pharyngeal postures for breathing and
decrease hyperfunction during phonation.
Voice therapy
The patient underwent three weekly sessions of voice and respi-
ratory retraining therapy. The respiratory retraining was aimed
at facilitating the most open laryngeal airway and improving the
patients perceived control over dyspneic episodes.
4,5
The
patient was trained to exhale for 4 seconds while producing
the voiceless fricative /s/ and then inhale through the mouth
with pursed lips. This manner of breathing was practiced
during rest and with mild exertion (stair climbing, walking).
Voice therapy focused on the use of resonant voice.
6
He was
trained to produce forward resonant voice characterized by
(1) sensation of anterior facial vibrations and (2) lack of self-
perceived tension in the throat or effort during phonation. These
techniques were practiced at the word, phrase, and, most impor-
tant, conversational level. Following therapy, the patient was
discharged from therapy and instructed to return to voice clinic
for re-evaluation.
Follow-up evaluations
The patient returned for follow-up appointments 3 and 6
months 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 VNS
was in a deactivation mode, revealed that the left vocal fold was
no longer immobile but showed evidence of hypomobility with
purposeful motion. The improvement in left vocal fold range of
motion remained stable in the subsequent two follow-up visits
when the device was off. However, after the VNS was turned
back on at lower settings, a decrease in vocal fold range of mo-
tion bilaterally during device activation was observed. This
nding indicated that vocal fold range of motion was affected
bilaterally 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 the
use of respiratory retraining therapy techniques. Use of the
same therapy techniques not only attenuated tonic symptoms
of dyspnea but reportedly were preventative to the extent that
the patient no longer experienced breathing attacks.
Implications
The current case presents the rst evidence of contralateral
adduction following VNS implantation resulting in dyspnea
and dysphonia persisting even throughout a period of VNS de-
activation. Three points require further investigation. First,
the contralateral adduction observed in this patient could have
been due to neural cross-innervation via the interarytenoid or
Galens anastomosis.
7,8
Ipsilateral adduction is expected even
at the lowest pulse duration levels (0.0250.50 mA) but
contralateral vocal fold adduction is not anticipated at such
low levels.
9
However, in the present case, the VNS was acti-
vated at large amplitudes (0.75 mA), which theoretically might
induce contralateral adduction due to bidirectional signal trans-
port not only directly to the left recurrent laryngeal nerve but
also from efferent brainstem reexes as a result of stimulation.
VNS stimulation of 10 Hz (the patients frequency setting) is
not expected to trigger adduction, especially of the contralateral
vocal fold.
10
TABLE 2.
VNS Setting and Subjective Patient Complaint at Each Time Point
Visit # Time VNS Settings Symptoms
1 Initial clinic visit Output: 1.0 mA
Frequency: 20 Hz
On: 30 s; off: 3 min
Severe dysphonia and dyspnea. Both
worsened with device on
2 3 mo posttherapy
1 wk post-VNS reactivation
Output: 0.25 mA
Frequency: 10 Hz
On: 14 s; off: 5 min
Dysphonia and dyspnea with VNSstimulation
and with exertion (absent stimulation)
3 6 mo posttherapy
3 mo post-VNS reactivation
Output: 0.75 mA
Frequency: 10 Hz
On: 14 s; off : 5 min
Dyspnea and dysphonia were 90% better
Telephone follow-up 13 mo posttherapy
10 mo post-VNS reactivation
Output: 0.75 mA
Frequency: 10 Hz
On: 14 s; off : 5 min
Awareness of symptoms but able to use
therapy 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 33
2 (3 mo post-tx) 28 26
3 (6 mo post-tx) 24 23
Journal of Voice, Vol. 28, No. 1, 2014 60
A second point of interest is that the persistence of the pa-
tients symptoms even after VNS deactivation may provide in-
sight to mechanisms of PVFM in patients following noxious
exposure. Some have hypothesized that central neuronal
changes occur with repeated noxious stimuli to the larynx, re-
sulting in PVFM even after cessation of the stimuli.
11
In this
case, the cyclic VNS activation triggering vocal fold adduction
may have caused neurologic changes at the central or peripheral
level resulting in ongoing laryngeal hyperadduction causing
dyspnea even after VNS deactivation.
A third point of interest relates to respiratory retraining as
a known effective treatment for dyspnea due to PVFM.
5,1214
Behavioral treatment of dyspnea caused by inappropriate
vocal fold adduction in relation to VNS activation has not
previously been reported in the literature. Typical treatments
for 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 successful
in alleviating symptoms of PVFM-like dyspnea in a patient
with VNS. Multidisciplinary teamwork of laryngology, neurol-
ogy, and speech-language pathology is critical in the manage-
ment of concomitant dyspnea and epilepsy.
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