Daytime Hypoxemia, Sleep-Disordered Breathing, and Laryngopharyngeal Findings in Multiple System Atrophy

Shimohata, Takayoshi; Shinoda, Hideo; Nakayama, Hideaki; Ozawa, Tetsutaro; Terajima, Kenshi; Yoshizawa, Hirohisa; Matsuzawa, Yoko; Onodera, Osamu; Naruse, Satoshi; Tanaka, Keiko; Takahashi, Sugata; Gejyo, Fumitake; Nishizawa, Masatoyo

doi:10.1001/archneur.64.6.856

Cited by 88 publications

(73 citation statements)

References 18 publications

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“…Four out of nine patients with severe sleepdisordered breathing (AHI 3 30) exhibited VCAP or CSA, which might be related to nocturnal sudden death in MSA [3][4][5]. The abnormalities in polysomnographic variables were independent of the duration of illness or UMSARS, a finding that is consistent with a previous report [26]. These findings suggest that critical sleep-related breathing problems including VCAP and CSA might emerge even as the primary presenting symptom of MSA [27][28][29].…”

Section: Discussionsupporting

confidence: 83%

The close relationship between life-threatening breathing disorders and urine storage dysfunction in multiple system atrophy

et al. 2010

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Survival of multiple system atrophy (MSA) depends on whether a variety of sleep-related breathing problems as well as autonomic failure (AF) occur. Since the brainstem lesions that cause respiratory and autonomic dysfunction overlap with each other, these critical manifestations might get worse in parallel. If so, the detection of AF, which is comparatively easy, might be predictive of a latent life-threatening breathing disorder. In 15 patients with MSA, we performed autonomic function tests composed of postural challenges and administered a questionnaire on bladder condition, as well as polysomnography and laryngoscopy during wakefulness and under anesthesia. Polysomnographic variables such as the apnea-hypopnea index (AHI) and oxygen saturation (SpO(2)) and the findings of laryngoscopy were compared with the degree of cardiac and urinary autonomic dysfunction. AHI, mean SpO(2) and the lowest SpO(2) showed significant correlations with urine storage dysfunction. In addition, patients with vocal cord abductor paralysis (VCAP) or central sleep apnea (CSA) contributing to nocturnal sudden death had more severe storage disorders than those without. On the other hand, no significant relationship between polysomnographic variables and orthostatic hypotension was observed except in the case of mean SpO(2). These results indicate that life-threatening breathing disorders have a close relationship with AF, and especially urine storage dysfunction. Therefore, longitudinal assessment of deterioration of the storage function might be useful for predicting the latent progress of VCAP and CSA.

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Section: Discussionsupporting

confidence: 83%

The close relationship between life-threatening breathing disorders and urine storage dysfunction in multiple system atrophy

et al. 2010

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“…Some studies demonstrate that CPAP may be ineffective in patients who have a severely floppy epiglottis because the pressure may further push the epiglottis into the laryngeal inlet. [24][25][26] The extent to which epiglottic collapse contributes to CPAP failure or non-adherence is not known and is an area of interest for future study. Although surgical resection or trimming of the epiglottis may be an option, no controlled studies have reported the efficacy of this approach, and the potential for side effects such as aspiration are not trivial.…”

Section: Discussionmentioning

confidence: 99%

Effect of Sleeping Position on Upper Airway Patency in Obstructive Sleep Apnea Is Determined by the Pharyngeal Structure Causing Collapse

Marques

Genta

Sands

et al. 2017

Sleep

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Objectives:In some patients, obstructive sleep apnea (OSA) can be resolved with improvement in pharyngeal patency by sleeping lateral rather than supine, possibly as gravitational effects on the tongue are relieved. Here we tested the hypothesis that the improvement in pharyngeal patency depends on the anatomical structure causing collapse, with patients with tongue-related obstruction and epiglottic collapse exhibiting preferential improvements. Methods: Twenty-four OSA patients underwent upper airway endoscopy during natural sleep to determine the pharyngeal structure associated with obstruction, with simultaneous recordings of airflow and pharyngeal pressure. Patients were grouped into three categories based on supine endoscopy: Tongue-related obstruction (posteriorly located tongue, N = 10), non-tongue related obstruction (collapse due to the palate or lateral walls, N = 8), and epiglottic collapse (N = 6). Improvement in pharyngeal obstruction was quantified using the change in peak inspiratory airflow and minute ventilation lateral versus supine. Results: Contrary to our hypothesis, patients with tongue-related obstruction showed no improvement in airflow, and the tongue remained posteriorly located while lateral. Patients without tongue involvement showed modest improvement in airflow (peak flow increased 0.07 L/s and ventilation increased 1.5 L/min). Epiglottic collapse was virtually abolished with lateral positioning and ventilation increased by 45% compared to supine position. Conclusions: Improvement in pharyngeal patency with sleeping position is structure specific, with profound improvements seen in patients with epiglottic collapse, modest effects in those without tongue involvement and-unexpectedly-no effect in those with tongue-related obstruction. Our data refute the notion that the tongue falls back into the airway during sleep via gravitational influences.

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“…■ Key words multiple system atrophy · sudden death · tracheostomy · continuous positive airway pressure · noninvasive positive pressure ventilation if they showed evidence for hereditary spinocerebellar ataxias by a molecular genetic test [10]. The onset of symptoms was determined as the time at which the initial symptoms of movement impairment were first noted by the individual and close family members.…”

Section: Methodsmentioning

confidence: 99%

Frequency of nocturnal sudden death in patients with multiple system atrophy

et al. 2008

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Sudden death has been reported in patients with multiple system atrophy (MSA), although the frequency of this event has not been well delineated. We investigated the frequency and potential causes of sudden death in patients with MSA. During the 5-year observation period, 10 of 45 patients with probable MSA died. The causes of death included sudden death of unknown etiology (seven patients), aspiration pneumonia (one patient), asphyxia after vomiting (one patient), and lung cancer (one patient). The mean survival time of patients with sudden death was 63.0 +/- 24.7 months (range, 39-116 months). Among seven patients who experienced sudden death, six were found to have died during sleep. Among these patients, two had been treated with tracheostomy and three with continuous positive airway pressure (CPAP) or noninvasive positive pressure ventilation (NPPV) during sleep, suggesting that these treatments do not always prevent sudden death in patients with MSA. Nocturnal sudden death should be recognized as the most common mechanism of death in patients with MSA.

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Daytime Hypoxemia, Sleep-Disordered Breathing, and Laryngopharyngeal Findings in Multiple System Atrophy

Cited by 88 publications

References 18 publications

The close relationship between life-threatening breathing disorders and urine storage dysfunction in multiple system atrophy

The close relationship between life-threatening breathing disorders and urine storage dysfunction in multiple system atrophy

Effect of Sleeping Position on Upper Airway Patency in Obstructive Sleep Apnea Is Determined by the Pharyngeal Structure Causing Collapse

Frequency of nocturnal sudden death in patients with multiple system atrophy

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