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Effects of long-term non-invasive ventilation on sleep structure in children with Spinal Muscular Atrophy type 2

Elisabetta Verrillo1, Martino Pavone1, Oliviero Bruni2

  • 1Sleep and Long-Term Ventilation Unit, Pediatric Pulmonology & Respiratory Intermediate Care Unit, Academic Department of Pediatrics (DPUO) Bambino Gesù Children's Hospital, IRCCS, Rome, Italy.

Sleep Medicine
|May 28, 2019
PubMed

Insights

Long-term non-invasive positive pressure ventilation (LTNPPV) in children with Spinal Muscular Atrophy type 2 (SMA2) only partially improves sleep arousability, with persistent differences in sleep architecture and microstructure compared to healthy controls.

Area of Science:

  • Pediatric Pulmonology
  • Sleep Medicine
  • Neuromuscular Disorders

Background:

  • Children with Spinal Muscular Atrophy type 2 (SMA2) exhibit altered sleep architecture, notably reduced arousability.
  • The impact of long-term ventilation on sleep in SMA2 is not well-documented.

Purpose of the Study:

  • To investigate the effects of long-term non-invasive positive pressure ventilation (LTNPPV) on sleep architecture in children with SMA2.
  • To compare sleep parameters in SMA2 patients on LTNPPV with age-matched controls.

Main Methods:

  • Nine children with SMA2 underwent polysomnography (PSG) during spontaneous breathing and after LTNPPV.
  • Sleep data were compared between the two conditions and with 15 age-matched healthy controls.

Main Results:

  • LTNPPV led to minor sleep architecture changes in SMA2 patients (increased N2%, decreased awakenings).
  • Significant differences persisted between SMA2 patients on LTNPPV and controls, including reduced total sleep time, awakenings, sleep efficiency, and REM sleep.
  • Cyclic alternating pattern (CAP) analysis revealed marginal changes with LTNPPV but significant differences between SMA2 patients and controls, with altered CAP subtype percentages.

Conclusions:

  • This study provides novel insights into sleep microstructure in pediatric SMA2 patients on LTNPPV.
  • LTNPPV offers partial improvement in arousability but does not fully normalize sleep in SMA2.
  • Persistent sleep microstructure alterations highlight the ongoing challenges in managing sleep in these patients.
Abstract

Keywords:
ArousabilityCyclic alternating patternLong term ventilationSleepSleep breathing disorderSpinal muscular atrophy type 2

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