Imaging and Surgical Findings in Patients with Hemi-Laryngopharyngeal Spasm and the Potential Role of MRI in the Diagnostic Work-Up

Avecillas-Chasín, Josué M; Kozoriz, Michael G.; Shewchuk, Jason R.; Heran, Manraj K. S.; Honey, Christopher R.

doi:10.3174/ajnr.a5851

Cited by 5 publications

(11 citation statements)

References 11 publications

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“…Our group has shown that the presence of a vascular conflict with the lower cranial nerves is common in the population. 3 This MRI finding is thus necessary but not sufficient for the diagnosis of HeLPS. Patients without a vascular conflict can be excluded from the diagnosis, but patients with this finding need further confirmatory tests.…”

Section: Discussionmentioning

confidence: 95%

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Hemi-laryngopharyngeal Spasm (HeLPS): Defining a New Clinical Entity

Morrison

Honey

2020

Ann Otol Rhinol Laryngol

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Objective: Hemi-laryngopharyngeal spasm (HeLPS) has recently been described in the neurosurgical literature as a cause of intermittent laryngopharyngeal spasm and cough due to vascular compression of the vagus nerve at the cerebellopontine angle. We present the diagnostic criteria for this syndrome. Methods: A retrospective chart review of six patients with HeLPS and three patients misdiagnosed with this condition are presented. All patients were diagnosed and treated at a tertiary care academic centre from July 2013 to July 2017. Results: Patients with HeLPS had five defining characteristics: 1) All patients had symptoms of episodic laryngopharyngeal spasm and coughing. Patients were asymptomatic between episodes and were refractory to speech therapy and reflux management. 2) Laryngoscopy showed hyperactive twitching of the ipsilateral vocal fold in two of the six patients. No other inter-episodic abnormalities were seen. 3) Botulinum toxin A injections into the thyroarytenoid muscle on the affected ipsilateral side reduced laryngopharyngeal spasms. Botulinum toxin injection in the contralateral thyroarytenoid muscle did not improve laryngopharyngeal spasm. 4) Magnetic resonance imaging revealed ipsilateral neurovascular compression of the vagus nerve rootlets by the posterior inferior cerebellar artery. 5) Microvascular decompression (MVD) surgery of the ipsilateral vagus nerve resolved all symptoms (follow-up 2-4 years). Conclusion: The diagnostic criteria for hemi-laryngopharyngeal spasm (HeLPS) are proposed. Otolaryngology recognition of this new clinical entity may lead to a surgical cure and avoid the unnecessary therapies associated with misdiagnosis. Level of Evidence: 4

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Section: Discussionmentioning

confidence: 95%

“…All patients had magnetic resonance imaging of the head using a CISS sequence to assess for neurovascular conflict between the vagus nerve rootlets and the posterior inferior cerebellar artery on the ipsilateral side of the symptoms (Figure 1). 3 All patients were followed for at least 2 years post-operatively.…”

Section: Methodsmentioning

confidence: 99%

Hemi-laryngopharyngeal Spasm (HeLPS): Defining a New Clinical Entity

Morrison

Honey

2020

Ann Otol Rhinol Laryngol

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“…Our previous MRI study reported that this incidental contact may be as high as 50% in asymptomatic individuals. 19 We are prospectively evaluating sequential unilateral proximal vagus nerve block as the diagnostic test of choice. Cessation of cough following a unilateral vagus nerve block ipsilateral but not contralateral to an MRI confirmed vascular compression may be the definitive test for VANCOUVER syndrome.…”

Section: Discussionmentioning

confidence: 99%

“…Our previous MRI study reported that this incidental contact may be as high as 50% in asymptomatic individuals. 19 We are prospectively evaluating sequential unilateral proximal vagus nerve block The looping posterior inferior cerebellar artery (large arrow) had its convex apex against the caudal rootlet of the left vagus nerve (thin arrow) and was encroaching on the nerve from anterior and inferior (as predicted from the MRI). The vessel has been moved slightly laterally, out from behind the nerve, in order to see it.…”

Section: 'A Vagal Neuralgia Could Manifest As a Sudden And Exaggeratementioning

confidence: 99%

Vagus Associated Neurogenic Cough Occurring Due to Unilateral Vascular Encroachment of Its Root: A Case Report and Proof of Concept of VANCOUVER Syndrome

Honey

Krüger

Morrison

et al. 2019

Ann Otol Rhinol Laryngol

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Objectives: A patient is presented with neurogenic cough due to a unilateral vascular compression of a vagus nerve rootlet at the brainstem with complete resolution of cough following microvascular decompression of that nerve. This etiology of a neurogenic cough has not been previously reported to our knowledge. The proportion of patients with neurogenic cough refractory to all current therapies and suffering with this treatable condition remains to be defined. We introduce the concept of Vagus Associated Neurogenic Cough Occurring due to Unilateral Vascular Encroachment of its Root (VANCOUVER syndrome) and present the salient features of this condition. Methods: A case review is presented with details of the patient’s history, examination, imaging, laryngoscopy, intraoperative findings, and long-term clinical outcome. Results: A 60-year-old man presented with a 15-year history of non-productive cough refractory to antibiotics, and anti-reflux medications. Investigations by an allergist, a cardiologist, a gastroenterologist, two pulmonologists, and an otolaryngologist were negative. MRI demonstrated a vascular compression of his left vagus nerve and microvascular decompression of that nerve resolved his symptoms. There were no surgical complications and the patient remains asymptomatic at 1 year. Conclusions: Neurogenic cough has been likened to a vagus nerve neuropathy in a similar way that trigeminal neuralgia is a trigeminal nerve neuropathy. Both cause intermittent sensory phenomena in their distribution and can be ameliorated with neuropathic medications. We demonstrate that neurogenic cough, like trigeminal neuralgia, may be caused by a vascular compression of its nerve root. A proposed mechanism of this type of neurogenic cough is presented along with a potential diagnostic paradigm for these patients.

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“…Honey et al proposed neurovascular compression of the vagus nerve rootlets identified on magnetic resonance imaging [ 21 ] as a potential cause of vagal dysfunction presenting in the larynx with sensory symptoms of abnormal throat sensations [ 22 ] associated with motor symptoms of laryngospasm/choking, neurogenic cough or intermittent stridor [ 23 ].…”

Section: Introductionmentioning

confidence: 99%

Supraglottic Botulinum Toxin Improves Symptoms in Patients with Laryngeal Sensory Dysfunction Manifesting as Abnormal Throat Sensation and/or Chronic Refractory Cough

Novakovic

Sheth

Stewart

et al. 2021

JCM

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Laryngeal sensory dysfunction (LSD) encompasses disorders of the vagal sensory pathways. Common manifestations include chronic refractory cough (CRC) and abnormal throat sensation (ATS). This study examined clinical characteristics and treatment outcomes of LSD using a novel approach of laryngeal supraglottic Onabotulinum toxin Type A injection (BTX). This was a retrospective review of clinical data and treatment outcomes of supraglottic BTX in patients with LSD. Between November 2019 and May 2021, 14 patients underwent 25 injection cycles of supraglottic BTX for treatment of symptoms related to LSD, including ATS and CRC. Primary outcome measures included the Newcastle Laryngeal Hypersensitivity Questionnaire (LHQ), Cough Severity Index (CSI), Reflux Symptom Index (RSI), and Voice Handicap Index-10 (VHI-10) at baseline and within three months of treatment. Pre- and post-treatment data were compared using a linear mixed model. After supraglottic BTX, LHQ scores improved by 2.6. RSI and CSI improved by 8.0 and 5.0, respectively. VHI-10 did not change as a result of treatment. Short-term response to SLN block was significantly associated with longer term response to BTX treatment. These findings suggest that LSD presents clinically as ATS and CRC along with other upper airway symptoms. Supraglottic BTX injection is a safe and effective technique in the treatment of symptoms of LSD.

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Imaging and Surgical Findings in Patients with Hemi-Laryngopharyngeal Spasm and the Potential Role of MRI in the Diagnostic Work-Up

Cited by 5 publications

References 11 publications

Hemi-laryngopharyngeal Spasm (HeLPS): Defining a New Clinical Entity

Hemi-laryngopharyngeal Spasm (HeLPS): Defining a New Clinical Entity

Vagus Associated Neurogenic Cough Occurring Due to Unilateral Vascular Encroachment of Its Root: A Case Report and Proof of Concept of VANCOUVER Syndrome

Supraglottic Botulinum Toxin Improves Symptoms in Patients with Laryngeal Sensory Dysfunction Manifesting as Abnormal Throat Sensation and/or Chronic Refractory Cough

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