An electrical cause of stridor: Pediatric vagal nerve stimulators

Kelts, Gregory; O’Connor, Peter; Hussey, Richard W.; Maturo, Stephen

doi:10.1016/j.ijporl.2014.10.037

Cited by 13 publications

(5 citation statements)

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“…[1], [2], [9], [11] Tonsillectomy and adenotonsillectomy are the first line of treatment for OSA in pediatric populations but has shown variable levels of success in VNS-induced sleep apnea. [11], [15], [17] This suggests that airway splint or positive airway pressure may not overcome the inhibitory action of vagal nerve on respiration in our subjects. Most of the respiratory events associated with VNS stimulation were hypopneas which were obstructive at least in subject 3 who had an intercostal EMG lead monitoring muscle activation.…”

Section: Discussionmentioning

confidence: 81%

“…Changing the VNS settings is a reasonable solution for VNS-induced SDB, although it must be balanced with seizure control. [1], [2], [9], [17] However, determining ideal VNS parameters for each individual subject while maintaining therapeutic levels of stimulation is challenging. Three of our subjects showed improvement in their VNS-induced SDB after titrations of their specific VNS settings.…”

Section: Discussionmentioning

confidence: 99%

“…While treatment of primary OSA with positive airway pressure in patients with epilepsy can reduce seizure frequency, [15] the association of VNS and therefore sleep disordered breathing (SDB) deserves a closer look, especially since several cases have been described in a recent review [16] and there is no established treatment. [1], [2], [17], [18] We report 4 subjects who experienced marked SDB during VNS activation and discuss the dilemma of VNS titration.…”

Section: Introductionmentioning

confidence: 98%

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Treatment of vagus nerve stimulator-induced sleep-disordered breathing: A case series

Johnson

Shah³

et al. 2019

Epilepsy & Behavior Reports

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Objective Vagus nerve stimulation (VNS) is a treatment option for patients with drug-resistant seizures, but it is also associated with sleep-disordered breathing (SDB). We present four patients with VNS who underwent polysomnography (PSG) concurrently with VNS stimulation monitoring and adjustment, and positive airway pressure (PAP) treatment. We demonstrate the importance of sleep apnea screening prior to VNS placement and the dilemma of optimizing VNS settings. Background VNS is a common adjunct therapy for refractory epilepsy. Despite its low side effect profile, complications of VNS include delayed arrhythmias, laryngopharyngeal dysfunction, obstructive sleep apnea, and tonsillar pain mimicking glossopharyngeal neuralgia. Risk of developing or exacerbating existing obstructive sleep apnea (OSA) limits the VNS settings, as there appears to be a dose dependent effect. OSA can further cause sleep fragmentation and cause hypoxia, potentially worsening seizures. Methods Four patients with drug-resistant epilepsy with VNS underwent PSG with concurrent VNS leads to monitor correlation of SDB and VNS. AHI was calculated to quantify SDB, and it was scored as non-VNS related when the VNS was off, and VNS-induced when the onset of SDB corresponded to VNS activation. Subsequent PAP and VNS adjustment was performed to treat the SDB episodes. Results Three out of four patients had non-VNS associated SDB, which improved with PAP treatment. All four patients had VNS-induced SDB episodes but none improved with PAP. The VNS-induced SDB events decreased in a dose dependent manner, when VNS was adjusted down and disappeared when turned off completely. Conclusion Our case series provides further evidence of VNS-induced SDB secondary to VNS. PAP treatment alone is ineffective for VNS-induced SDB. Screening for OSA before VNS implant is crucial; further research is needed to establish optimal VNS parameters for prevention andminimization of VNS-induced SDB along with other possible treatments.

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Section: Discussionmentioning

confidence: 81%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 98%

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Treatment of vagus nerve stimulator-induced sleep-disordered breathing: A case series

Johnson

Shah³

et al. 2019

Epilepsy & Behavior Reports

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“…Our cases demonstrate that other sleep disorders, such as stridor and catathrenia, may also be a complication of VNS treatment. Stridor, as a side effect of VNS therapy, has been already reported in two adults (Bhatt et al ., 2010; St Louis and Faber, 2010) and two paediatric (Kelts et al ., 2015) epileptic patients, and its resolution was seen in all patients following adjustment of VNS settings. Nonetheless, to the best of our knowledge, VNS‐induced catathrenia has not previously been described.…”

Section: Discussionmentioning

confidence: 90%

Complex sleep‐disordered breathing after vagus nerve stimulation: broadening the spectrum of adverse events of special interest

Santos¹,

Bentes²,

Teodoro³

et al. 2020

Epileptic Disorders

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Two young males with refractory epilepsy of unknown aetiology were referred for vagus nerve stimulation (VNS). Sleep disturbances emerged following VNS parameter changes. In Patient 1, video‐polysomnogram (PSG) disclosed snoring and catathrenia in non‐REM sleep. Central apnoea also occurred, but more rarely. In Patient 2, video‐PSG showed mixed apnoea with desaturation and episodes of stridor followed by a catathrenia‐like sound. A drug‐induced sleep endoscopy (DISE) revealed, during VNS OFF time, glossoptosis, “trap door” of the epiglottis, and paresis of the left side of the larynx and ipsilateral vocal cords. During ON time, there were periods of pharyngeal collapse, in which video‐PSG revealed patterns suggestive of both obstructive and central sleep apnoea. All these sleep‐related phenomena were coincident with VNS ON time. In the first patient, VNS parameter adjustment was sufficient to successfully reverse all the symptoms, whereas the other patient required concomitant treatment with continuous positive airway pressure. The data broaden our knowledge about sleep disorders related to VNS, in particular stridor and catathrenia. We suggest that central sleep apnoea may be associated with laryngeal occlusion. DISE may be considered in selected cases as a valuable clinical tool to evaluate, in a single session, the effectiveness of multiple VNS parameter changes on respiration and laryngeal side effects. [Published with video sequences].

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“…Therefore, these studies and other data 84‐88 indicate that VNS effects on vocal function remains as a prominent area‐of‐concern. In addition, vagus nerve stimulation can also be a cause of stridor in a pediatric population with epilepsy 89 . Other issues like appropriate electrode size is an important consideration during surgical implantation, so that compression injury to the VN is avoided 90 .…”

Section: Discussionmentioning

confidence: 99%

Vagus nerve stimulation paired with tones for tinnitus suppression: Effects on voice and hearing

Kochilas

Cacace

Arnold

et al. 2020

Laryngoscope Investig Oto

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Objective: In individuals with chronic tinnitus, our interest was to determine whether daily low-level electrical stimulation of the vagus nerve paired with tones (paired-VNSt) for tinnitus suppression had any adverse effects on motor-speech production and physiological acoustics of sustained vowels. Similarly, we were also interested in evaluating for changes in pure-tone thresholds, word-recognition performance, and minimum-masking levels.Both voice and hearing functions were measured repeatedly over a period of 1 year. Study design: Longitudinal with repeated-measures.Methods: Digitized samples of sustained frontal, midline, and back vowels (/e/, /o/, /ah/) were analyzed with computer software to quantify the degree of jitter, shimmer, and harmonic-to-noise ratio contained in these waveforms. Pure-tone thresholds, monosyllabic word-recognition performance, and MMLs were also evaluated for VNS alterations. Linear-regression analysis was the benchmark statistic used to document change over time in voice and hearing status from a baseline condition.Results: Most of the regression functions for the vocal samples and audiometric variables had slope values that were not significantly different from zero. Four of the nine vocal functions showed a significant improvement over time, whereas three of the pure tone regression functions at 2-4 kHz showed some degree of decline; all changes observed were for the left ear, all were at adjacent frequencies, and all were ipsilateral to the side of VNS. However, mean pure-tone threshold changes did not exceed 4.29 dB from baseline and therefore, would not be considered clinically significant. In some individuals, larger threshold shifts were observed. No significant regression/slope effects were observed for word-recognition or MMLs.Meeting information: Portions of this study were presented at

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An electrical cause of stridor: Pediatric vagal nerve stimulators

Cited by 13 publications

References 14 publications

Treatment of vagus nerve stimulator-induced sleep-disordered breathing: A case series

Treatment of vagus nerve stimulator-induced sleep-disordered breathing: A case series

Complex sleep‐disordered breathing after vagus nerve stimulation: broadening the spectrum of adverse events of special interest

Vagus nerve stimulation paired with tones for tinnitus suppression: Effects on voice and hearing

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