Unexpectedly severe sleep and respiratory pathology in patients with amyotrophic lateral sclerosis
Open accessAbstract
Daytime fatigue and sleep disturbance are frequent complaints in patients with amyotrophic lateral sclerosis (ALS). However, polysomnographic data are sparse. Nocturnal respiratory insufficiency may occur despite nearly normal daytime pulmonary function. We describe the clinical presentation and polysomnographic findings in two patients with clinically and electrophysiologically confirmed ALS with minimal weakness but excessive daytime sleepiness. Polysomnography in the first patient showed a respiratory disturbance index of 43.5, and profound oxygen desaturations to 62%. The second patient had prolonged periods of hypoventilation, with oxygen saturations oscillating between 86 and 83%. Both patients showed severe sleep maintenance insomnia with a sleep efficiency < 40% and frequent arousals while asleep. Application of continuous positive airway pressure (CPAP) restored normal nocturnal ventilation, blood oxygenation and sleep parameters in the first patient; compliance, however, was poor. The second patient was unable to tolerate CPAP. We conclude that ALS patients with excessive daytime sleepiness or insomnia should undergo polysomnography to adequately diagnose nocturnal respiratory insufficiency and sleep disturbance. Compliance with treatment, however, may be poor.
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Barthlen and colleagues were among the first to describe the association of insomnia symptoms and reduced CPAP adherence among two patients with severe OSA and insomnia symptoms.
“Barthlen and colleagues were among the first to describe the association of insomnia symptoms and reduced CPAP adherence among two patients with severe OSA and insomnia symptoms [68].”
From Co-morbid insomnia and sleep apnea (COMISA): Prevalence, consequences, methodological considerations, and recent randomized controlled trials (2019) · cites this paper as [68]
The Abstract and case reports describe two patients with severe sleep-maintenance insomnia and poor CPAP compliance or tolerance. But they were patients with ALS, not two patients with severe obstructive sleep apnea: the Discussion says frank obstructive apneas were absent, and Patient 2 had no apneas. The paper reports these findings together, rather than establishing an association between insomnia symptoms and reduced CPAP adherence. (LLM, conf=0.89)
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References 8 · Cited by 53
Research works printed but not linked: 1
- Phillips L, Kinnear W, Scriven N, Orpe V, Jefferson D (1997). Predictors of hypercapnic respiratory failure in patients with motor neurone disease. Proceedings of the 8th International Symposion on ALS/MND, November 3]5. Glasgow, UK, p.
- Authors
- Barthlen, G. M., & Lange, D. J.
- Affiliations
- Mount Sinai Hospital (US)
- Corresponding author
- Lange, D. J.
- Venue
- European Journal of Neurology
- Publisher
- Wiley
- Published
- 2000-06
- Volume
- 7
- Issue
- 3
- Pages
- 299-302
- ISSN
- 1351-5101 (ISSN-L)
- 1468-1331
- OpenAlex
- W2040615884
- Cited by
- 53
- References
- 8
- Citations by year
- 2026: 1 · 2024: 2 · 2023: 1 · 2022: 3 · 2021: 1 · 2020: 1 · 2019: 3 · 2016: 4 · 2015: 2 · 2014: 5 · 2013: 1 · 2012: 5
- FWCI
- 0.61
- Type
- journal article
- Language
- en
- Open access
- bronze
- doi.org
- Locations
- European Journal of Neurology (publishedVersion)
- PubMed (publishedVersion)
- Topics
- Health Sciences › Medicine › Physiology › Obstructive Sleep Apnea Research
- Health Sciences › Medicine › Neurology › Amyotrophic Lateral Sclerosis Research
- Social Sciences › Psychology › Experimental and Cognitive Psychology › Sleep and related disorders
- Keywords
- amyotrophic lateral sclerosis, excessive daytime sleepiness, sleep maintenance insomnia, polysomnography findings, hypoventilation, oxygen desaturation, continuous positive airway pressure, CPAP adherence, arousal
- MeSH terms
- Amyotrophic Lateral Sclerosis, Arousal, Circadian Rhythm, Humans, Hypoventilation, Sleep Initiation and Maintenance Disorders, Male, Middle Aged, Oxygen, Positive-Pressure Respiration, Respiration Disorders, Sleep, Sleep Apnea Syndromes, Sleep Wake Disorders, Sleep Stages, Polysomnography
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