title: Effect of sleep stage on breathing in children with central hypoventilation.
abstract: The early literature suggests that hypoventilation in infants with congenital central hypoventilation syndrome (CHS) is less severe during rapid eye movement (REM) than during non-REM (NREM) sleep. However, this supposition has not been rigorously tested, and subjects older than infancy have not been studied. Given the differences in anatomy, physiology, and REM sleep distribution between infants and older children, and the reduced number of limb movements during REM sleep, we hypothesized that older subjects with CHS would have more severe hypoventilation during REM than NREM sleep. Nine subjects with CHS, aged (mean +/- SD) 13 +/- 7 yr, were studied. Spontaneous ventilation was evaluated by briefly disconnecting the ventilator under controlled circumstances. Arousal was common, occurring in 46% of REM vs. 38% of NREM trials [not significant (NS)]. Central apnea occurred during 31% of REM and 54% of NREM trials (NS). Although minute ventilation declined precipitously during both REM and NREM trials, hypoventilation was less severe during REM (drop in minute ventilation of 65 +/- 23%) than NREM (drop of 87 +/- 16%, P = 0.036). Despite large changes in gas exchange during trials, there was no significant change in heart rate during either REM or NREM sleep. We conclude that older patients with CHS frequently have arousal and central apnea, in addition to hypoventilation, when breathing spontaneously during sleep. The hypoventilation in CHS is more severe during NREM than REM sleep. We speculate that this may be due to increased excitatory inputs to the respiratory system during REM sleep.
authors.affiliation: Sleep Center, The Children's Hospital of Philadelphia, University of Pennsylvania School of Medicine, Philadelphia, Pennsylvania 19104, USA.
authors.forename: Jingtao
authors.initials: J
authors.lastname: Huang
authors.forename: Ian M
authors.initials: IM
authors.lastname: Colrain
authors.forename: Howard B
authors.initials: HB
authors.lastname: Panitch
authors.forename: Ignacio E
authors.initials: IE
authors.lastname: Tapia
authors.forename: Michael S
authors.initials: MS
authors.lastname: Schwartz
authors.forename: John
authors.initials: J
authors.lastname: Samuel
authors.forename: Michelle
authors.initials: M
authors.lastname: Pepe
authors.forename: Preetam
authors.initials: P
authors.lastname: Bandla
authors.forename: Ruth
authors.initials: R
authors.lastname: Bradford
authors.forename: Yael P
authors.initials: YP
authors.lastname: Mosse
authors.forename: John M
authors.initials: JM
authors.lastname: Maris
authors.forename: Carole L
authors.initials: CL
authors.lastname: Marcus
mesh_terms: D000293:Adolescent; D000328:Adult; D002648:Child; D002675:Child, Preschool; D004247:DNA / Q000096:biosynthesis / Q000235:genetics; D003627:Data Interpretation, Statistical; D005260:Female; D018398:Homeodomain Proteins / Q000235:genetics; D006801:Humans; D006935:Hypercapnia / Q000503:physiopathology; D008297:Male; D009068:Movement / Q000502:physiology; D017286:Polysomnography; D011659:Pulmonary Gas Exchange / Q000502:physiology; D015656:Respiratory Mechanics / Q000502:physiology*; D012890:Sleep / Q000502:physiology; D020182:Sleep Apnea, Central / Q000503:physiopathology*; D012894:Sleep Stages / Q000502:physiology*; D012895:Sleep, REM / Q000502:physiology; D014157:Transcription Factors / Q000235:genetics
chemical_list: D018398:Homeodomain Proteins; C109220:NBPhox protein; D014157:Transcription Factors; D004247:DNA