Effect of Laser Arytenoidectomy on Respiratory Stridor Caused by Multiple System Atrophy

Chitose, Shun‐ichi; Kikuchi, Atsushi; Ikezono, Keiko; Umeno, Hirohito; Nakashima, Tadashi

doi:10.5664/jcsm.2272

Cited by 12 publications

(9 citation statements)

References 8 publications

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“…In addition, we performed a literature review and searched PubMed using the terms "multiple system atrophy" OR "Shy‐Drager Syndrome" OR "olivo‐ponto‐cerebellar atrophy” OR "striato‐nigral degeneration" AND “larynx" and identified 52 relevant articles between 1979 and 2020, with 44 case reports 11,13,19‐39 or case series 9,14,40‐57 (total number of patients 232), 10 studies 12,17,56,58‐64 (total number of patients 328), and 2 reviews 65,66 …”

Section: Discussionmentioning

confidence: 99%

Laryngeal Movement Disorders in Multiple System Atrophy: A Diagnostic Biomarker?

et al. 2020

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A BS TRACT: Background: Multiple system atrophy (MSA) is a rare neurodegenerative disorder, and its parkinsonian variant can be difficult to delineate from Parkinson's disease (PD). Despite laryngeal dysfunction being associated with decreased life expectancy and quality of life, systematic assessments of laryngeal dysfunction in large cohorts are missing. Objectives: The objective of this study was to systematically assess laryngeal dysfunction in MSA and PD and identify laryngeal symptoms that allow for differentiating MSA from PD. Methods: Patients with probable or possible MSA underwent flexible endoscopic evaluation of swallowing performing a systematic task protocol. Findings were compared with an age-matched PD cohort. Results: A total of 57 patients with MSA (64 [59-71] years; 35 women) were included, and task assessments during endoscopic examination compared with 57 patients with PD (67 [60-73]; 28 women). Patients with MSA had a shorter disease duration (4 [3-5] years vs 7 [5-10]; P < 0.0001) and higher disease severity (Hoehn & Yahr stage 4 [3-4] vs 3 [2-4]; P < 0.0001). Of the patients with MSA, 43.9% showed clinically overt laryngeal dysfunction with inspiratory stridor. During endoscopic task assessment, however, 93% of patients with MSA demonstrated laryngeal dysfunction in contrast with only 1.8% of patients with PD (P < 0.0001). Irregular arytenoid cartilages movements were present in 91.2% of patients with MSA, but in no patients with PD (P < 0.0001). Further findings included vocal fold motion impairment (75.4%), paradoxical vocal fold motion (33.3%), and vocal fold fixation (19.3%). One patient with PD showed vocal fold motion impairment. Conclusion: Laryngeal movement disorders are highly prevalent in patients with MSA when assessed by a specific task protocol despite the lack of overt clinical symptoms. Our data suggest that irregular arytenoid cartilage movements could be used as a clinical marker to delineate MSA from PD with a specificity of 1.0 and sensitivity 0.9.

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Section: Discussionmentioning

confidence: 99%

Laryngeal Movement Disorders in Multiple System Atrophy: A Diagnostic Biomarker?

et al. 2020

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“…In MSA patients however, it must be taken into account that the adductor muscles are still activated and exhibit force on the suture providing the laterofixation (31). Some MSA patients underwent arytenoidectomy with conflicting outcomes (30, 55).…”

Section: Laryngeal Findings In Msamentioning

confidence: 99%

The Shaking Palsy of the Larynx—Potential Biomarker for Multiple System Atrophy: A Pilot Study and Literature Review

et al. 2019

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In its early stages multiple system atrophy (MSA), a neurodegenerative movement disorder, can be difficult to differentiate from idiopathic Parkinson's disease (PD), and emphasis has been put on identifying premotor symptoms to allow for its early identification. The occurrence of vegetative symptoms in addition to motor impairment, such as orthostatic hypotension and neurogenic bladder dysfunction, enable the clinical diagnosis in the advanced stages of the disease. Usually with further disease progression, laryngeal abnormalities become clinically evident and can manifest in laryngeal stridor due to impaired vocal fold motion, such as vocal fold abduction restriction, mostly referred to as vocal fold paresis, or paradoxical vocal fold adduction during inspiration. While the pathogenesis of laryngeal stridor is discussed controversially, its occurrence is clearly associated with reduced life expectancy. Before the clinical manifestation of laryngeal dysfunction however, abnormal vocal fold motion can already be seen in patients that might not yet fulfill the diagnostic criteria of MSA. In this article we summarize the current literature on pharyngolaryngeal findings in MSA and report preliminary findings from a pilot study investigating eight consecutive MSA patients. Patients showed varying speech abnormalities. Only 2/8 patients exhibited laryngeal stridor. However, during FEES, all patients presented with irregular arytenoid cartilages movements and vocal fold abduction restriction. 3/8 showed vocal fold fixation and 1/8 paradoxical vocal fold motion. All patients presented with oropharyngeal dysphagia, 5/8 with penetration or aspiration events. We suggest that specific abnormal vocal fold motion can help identifying MSA patients and may allow for delimiting this disorder from idiopathic PD. These findings therefore may serve as a novel clinical biomarker for MSA. Based on the available data and our preliminary clinical experience we developed a standardized easy-to-implement task-protocol to be performed during flexible endoscopic evaluation of swallowing (FEES) for detection of MSA-related pharyngolaryngeal movement disorders. Furthermore, we initiated a prospective study to evaluate the diagnostic utility of this protocol.

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“…Single case reports have described the use of posterior cordotomy and arytenoidectomy, 28,43,44,55 and botulinum toxin relieved dystonic stridor in 3 of 4 patients 1 month after inoculation. 45…”

Section: Resultsmentioning

confidence: 99%

“…Statement is based on core literature consisting of Class IV level studies including case reports. 28,43–45,55 There is insufficient evidence for minimally invasive procedures and botulinum toxin injections for the symptomatic treatment of stridor.…”

Section: Resultsmentioning

confidence: 99%

Stridor in multiple system atrophy

et al. 2019

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Multiple system atrophy (MSA) is a neurodegenerative disorder characterized by a combination of autonomic failure, cerebellar ataxia, and parkinsonism. Laryngeal stridor is an additional feature for MSA diagnosis, showing a high diagnostic positive predictive value, and its early occurrence might contribute to shorten survival. A consensus definition of stridor in MSA is lacking, and disagreement persists about its diagnosis, prognosis, and treatment. An International Consensus Conference among experts with methodological support was convened in Bologna in 2017 to define stridor in MSA and to reach consensus statements for the diagnosis, prognosis, and treatment. Stridor was defined as a strained, high-pitched, harsh respiratory sound, mainly inspiratory, occurring only during sleep or during both sleep and wakefulness, and caused by laryngeal dysfunction leading to narrowing of the rima glottidis. According to the consensus, stridor may be recognized clinically by the physician if present at the time of examination, with the help of a witness, or by listening to an audio recording. Laryngoscopy is suggested to exclude mechanical lesions or functional vocal cord abnormalities related to different neurologic conditions. If the suspicion of stridor needs confirmation, drug-induced sleep endoscopy or video polysomnography may be useful. The impact of stridor on survival and quality of life remains uncertain. Continuous positive airway pressure and tracheostomy are both suggested as symptomatic treatment of stridor, but whether they improve survival is uncertain. Several research gaps emerged involving diagnosis, prognosis, and treatment. Unmet needs for research were identified.

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Effect of Laser Arytenoidectomy on Respiratory Stridor Caused by Multiple System Atrophy

Cited by 12 publications

References 8 publications

Laryngeal Movement Disorders in Multiple System Atrophy: A Diagnostic Biomarker?

Laryngeal Movement Disorders in Multiple System Atrophy: A Diagnostic Biomarker?

The Shaking Palsy of the Larynx—Potential Biomarker for Multiple System Atrophy: A Pilot Study and Literature Review

Stridor in multiple system atrophy

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