Tremulous arytenoid movements predict severity of glottic stenosis in multiple system atrophy

Ozawa, Tetsutaro; Shinoda, Hideo; Tomita, Masahiko; Shimohata, Takayoshi; Nakayama, Hideaki; Nishizawa, Masatoyo

doi:10.1002/mds.23090

Cited by 15 publications

(18 citation statements)

References 27 publications

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“…Irregular ACM at rest were present in 75% of patients. These findings are in line with previous results from laryngoscopy studies that found irregular ACM in 50–100% of cases (27, 36–38). Irregular involuntary movements have been described in MSA patients to occur predominantly in hands and fingers.…”

Section: Discussionsupporting

confidence: 93%

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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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Section: Discussionsupporting

confidence: 93%

“…Ozawa and colleagues performed fiberendoscopic laryngoscopy on 28 MSA patients and 14 healthy controls. In 18 (64.3%) MSA patients irregular tremulous movement of the arytenoid cartilages was detected, none in the healthy control group (38).…”

Section: Laryngeal Findings In Msamentioning

confidence: 97%

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 addition, we performed a literature review and searched PubMed using the terms "multiple system atrophy" OR "Shy-Drager Syndrome" OR "olivoponto-cerebellar atrophy" OR "striato-nigral degeneration" AND "larynx" and identified 52 relevant articles between 1979 and 2020, with 44 case reports 11,13, or case series 9,14,[40][41][42][43][44][45][46][47][48][49][50][51][52][53][54][55][56][57] (total number of patients 232), 10 studies 12,17,56,[58][59][60][61][62][63][64] (total number of patients 328), and 2 reviews. 65,66 In summary, the case reports/series assessed the incidence of single laryngeal findings in patients with MSA either observed clinically during endoscopy or laryngeal electromyography.…”

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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“…A previous study with laryngoscopy and video monitoring suggested that the presence of tremulous arytenoid movement was associated with the severity of glottis stenosis, and that this is clearly correlated with disease duration in MSA patients [ 27 ]. The occurrence of rhythmic waveform could be another marker of severe glottis stenosis.…”

Section: Discussionmentioning

confidence: 99%

“…Although the pathogenic mechanisms underlying MSA stridor are still controversial, laryngeal abductor weakness or laryngeal adductor dystonia are considered a cause of stridor in MSA patients [ 28 ]. Neurogenic atrophy of the posterior cricoarytenoid muscle suggested the laryngeal abductor weakness, whereas dysfunction in the inhibitory brainstem autonomic pathways may cause dystonia in the laryngeal adductor muscles [ 27 – 29 ]. In this study, patients who displayed rhythmic signals had a worse outcome than those exhibiting less rhythmic waveform.…”

Section: Discussionmentioning

confidence: 99%

Acoustic Characteristics of Stridor in Multiple System Atrophy

et al. 2016

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Nocturnal stridor is a breathing disorder prevalent in patients with multiple system atrophy (MSA). An improved understanding of this breathing disorder is essential since nocturnal stridor carries a poor prognosis (an increased risk of sudden death). In this study, we aimed to classify types of stridor by sound analysis and to reveal their clinical significance. Patients who met the criteria for probable MSA and had undergone polysomnography (PSG) were recruited. Patients were then assessed clinically with sleep questionnaires, including the Pittsburgh Sleep Quality Index, and the Hoehn and Yahr scale. Nocturnal stridor and snoring were analyzed with the Multi-Dimensional Voice Program. Nocturnal stridor was recorded in 22 patients and snoring in 18 patients using the PSG. Waveforms of stridors were classified into rhythmic or semirhythmic after analysis of the oscillogram. Formants and harmonics were observed in both types of stridor, but not in snoring. Of the 22 patients diagnosed with stridor during the present study, fifteen have subsequently died, with the time to death after the PSG study being 1.9 ± 1.4 years (range 0.8 to 5.0 years). The rhythmic waveform group presented higher scores on the Hoehn and Yahr scale and the survival outcome of this group was lower compared to the semirhythmic waveform group (p = 0.030, p = 0.014). In the Kaplan Meier’s survival curve, the outcome of patients with rhythmic waveform was significantly less favorable than the outcome of patients with semirhythmic waveform (log-rank test, p < 0.001). Stridor in MSA can be classified into rhythmic and semirhythmic types and the rhythmic component signifies a poorer outcome.

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Tremulous arytenoid movements predict severity of glottic stenosis in multiple system atrophy

Cited by 15 publications

References 27 publications

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

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

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

Acoustic Characteristics of Stridor in Multiple System Atrophy

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