OBJECTIVES: To determine the prevalence and type of sleep-disordered breathing among patients with Prader-Willi syndrome (PWS) and its relationship to such neurobehavioral abnormalities as mental retardation, obsessive-compulsive behavior, and conduct disorders. STUDY DESIGN: Polysomnography (PSG) studies were conducted in 13 unselected subjects with PWS (age 1.5 to 28 years). PSG results were compared with tests of behavior and cognition (Development Behavior Checklist [DBC], Auditory Continuous Performance Test [ACPT], and Wechsler Intelligence Scale appropriate for age). RESULTS: Nine of 13 (69%) subjects had > 10 apneas and hypopneas per hour of sleep. Apart from a 2-year-old subject with normal body weight who demonstrated severe central hypopnea in rapid eye movement sleep, the sleep-breathing disturbance was due to upper airway obstruction. Age-adjusted body mass index was associated with more severe hypoxemia during sleep (min SaO2, r = -.87, P < .005) and more sleep disruption (arousals/hour of sleep, r = .62, P < .05; sleep efficiency, r = -.66, P < .05). Increasing severity of obstructive sleep apnea (OSA) or sleep disturbance was associated with daytime inactivity/sleepiness and autistic-relating behavior (DBC) and with impulsiveness (ACPT). Unexpectedly, sleep hypoxemia appeared to be predictive of increased performance IQ. CONCLUSIONS: OSA is prevalent among subjects with PWS and is associated with increased body mass, daytime inactivity/ sleepiness, and some behavioral disturbances.
OBJECTIVES: To determine the prevalence and type of sleep-disordered breathing among patients with Prader-Willi syndrome (PWS) and its relationship to such neurobehavioral abnormalities as mental retardation, obsessive-compulsive behavior, and conduct disorders. STUDY DESIGN: Polysomnography (PSG) studies were conducted in 13 unselected subjects with PWS (age 1.5 to 28 years). PSG results were compared with tests of behavior and cognition (Development Behavior Checklist [DBC], Auditory Continuous Performance Test [ACPT], and Wechsler Intelligence Scale appropriate for age). RESULTS: Nine of 13 (69%) subjects had > 10 apneas and hypopneas per hour of sleep. Apart from a 2-year-old subject with normal body weight who demonstrated severe central hypopnea in rapid eye movement sleep, the sleep-breathing disturbance was due to upper airway obstruction. Age-adjusted body mass index was associated with more severe hypoxemia during sleep (min SaO2, r = -.87, P < .005) and more sleep disruption (arousals/hour of sleep, r = .62, P < .05; sleep efficiency, r = -.66, P < .05). Increasing severity of obstructive sleep apnea (OSA) or sleep disturbance was associated with daytime inactivity/sleepiness and autistic-relating behavior (DBC) and with impulsiveness (ACPT). Unexpectedly, sleep hypoxemia appeared to be predictive of increased performance IQ. CONCLUSIONS: OSA is prevalent among subjects with PWS and is associated with increased body mass, daytime inactivity/ sleepiness, and some behavioral disturbances.
Authors: Jessica Mackay; Gillian M Nixon; Antony R Lafferty; Geoff Ambler; Nitin Kapur; Philip B Bergman; Cara Schofield; Chris Seton; Andrew Tai; Elaine Tham; Komal Vora; Patricia Crock; Charles Verge; Yassmin Musthaffa; Greg Blecher; Daan Caudri; Helen Leonard; Peter Jacoby; Andrew Wilson; Catherine S Choong; Jenny Downs Journal: J Autism Dev Disord Date: 2021-09-08
Authors: Jessica Duis; Lara C Pullen; Maria Picone; Norman Friedman; Stephen Hawkins; Elise Sannar; Anna C Pfalzer; Althea Robinson Shelton; Deepan Singh; Phyllis C Zee; Daniel G Glaze; Amee Revana Journal: J Clin Sleep Med Date: 2022-06-01 Impact factor: 4.324
Authors: Korwyn Williams; Ann Scheimann; Vernon Sutton; Elizabeth Hayslett; Daniel G Glaze Journal: J Clin Sleep Med Date: 2008-04-15 Impact factor: 4.062