Literature DB >> 8040765

Polysomnographic characteristics of patients with Rett syndrome.

C L Marcus1, J L Carroll, S A McColley, G M Loughlin, S Curtis, P Pyzik, S Naidu.   

Abstract

During wakefulness, patients with Rett syndrome have disordered breathing. To understand further this ventilatory control disorder, we performed polysomnography in 30 patients with Rett syndrome and 30 control subjects (female subjects with primary snoring). The median age was 7 years (range, 1 to 32 years) for Rett syndrome and 6 years (range, 1 to 17 years) for control subjects. During periods of wakefulness, 67% of patients with Rett syndrome had the characteristic pattern of disordered breathing (i.e., episodes of hyperventilation followed by central apnea and desaturation). No such events occurred during sleep. Sleep efficiency and sleep architecture were similar for both groups. During sleep, there was no difference in duration of periodic breathing, number of episodes of central apnea with desaturation, or number of episodes of obstructive apnea or end-tidal carbon dioxide tension between the two groups. Although arterial oxygen saturation during rapid eye movement (REM) sleep was slightly lower in patients with Rett syndrome (nadir, 94% +/- 2% vs 96% +/- 2%), it remained within the normal range. Parental history reflected the awake respiratory findings in most cases. We conclude that patients with Rett syndrome have normal breathing during non-rapid eye movement (NREM) sleep. We speculate that patients with Rett syndrome have normal brain-stem control of ventilation, and that the disordered breathing seen during wakefulness is due to an abnormality of the cortical influence on ventilation.

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Year:  1994        PMID: 8040765     DOI: 10.1016/s0022-3476(94)70196-2

Source DB:  PubMed          Journal:  J Pediatr        ISSN: 0022-3476            Impact factor:   4.406


  31 in total

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Review 2.  Breathing dysfunction in Rett syndrome: understanding epigenetic regulation of the respiratory network.

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3.  The course of awake breathing disturbances across the lifespan in Rett syndrome.

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Journal:  Brain Dev       Date:  2018-04-12       Impact factor: 1.961

4.  Central sleep apnoea in Rett syndrome.

Authors:  Giuseppe d'Orsi; Vincenzo Demaio; Francesco Scarpelli; Teresa Calvario; Mauro G Minervini
Journal:  Neurol Sci       Date:  2009-06-25       Impact factor: 3.307

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7.  Polysomnographic findings in Rett syndrome: a case-control study.

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Journal:  Sleep Breath       Date:  2012-03-07       Impact factor: 2.816

Review 8.  Breathing abnormalities in animal models of Rett syndrome a female neurogenetic disorder.

Authors:  Chun Jiang; Ningren Cui; Weiwei Zhong; Christopher M Johnson; Yang Wu
Journal:  Respir Physiol Neurobiol       Date:  2016-11-21       Impact factor: 1.931

9.  Mecp2 Disruption in Rats Causes Reshaping in Firing Activity and Patterns of Brainstem Respiratory Neurons.

Authors:  Yang Wu; Ningren Cui; Hao Xing; Weiwei Zhong; Colin Arrowood; Christopher M Johnson; Chun Jiang
Journal:  Neuroscience       Date:  2018-11-17       Impact factor: 3.590

10.  Dream enactment behavior: review for the clinician.

Authors:  Marc Baltzan; Chun Yao; Dorrie Rizzo; Ron Postuma
Journal:  J Clin Sleep Med       Date:  2020-11-15       Impact factor: 4.062

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