Sleep cycling alternating pattern (CAP) expression is associated with hypersomnia and GH secretory pattern in Prader-Willi syndrome

Lorenzo Priano, Graziano Grugni, Giacinta Miscio, Giulia Guastamacchia, Lorenzo Toffolet, Alessandro Sartorio, Alessandro Mauro

Research output: Contribution to journalArticle

Abstract

Background and purpose: Hypersomnia, sleep-disordered breathing and narcoleptic traits such as rapid eye movement (REM) sleep onset periods (SOREMPs) have been reported in Prader-Willi syndrome (PWS). In a group of young adult patients with genetically confirmed PWS we evaluated sleep and breathing polysomnographically, including cycling alternating pattern (CAP), and we analyzed the potential interacting role of sleep variables, sleep-related breathing abnormalities, hypersomnia, severity of illness variables and growth hormone (GH) secretory pattern. Patients and methods: Eleven males and 7 females (mean age: 27.5±5.5 years) were submitted to a full night of complete polysomnography and the multiple sleep latency test (MSLT). GH secretory pattern was evaluated by a standard GH-releasing hormone plus arginine test. Sixteen non-obese healthy subjects without sleep disturbances were recruited as controls. Results: Compared to controls PWS patients showed reduced mean MSLT score (P <0.001), reduced mean latency of sleep (P = 0.03), increased REM sleep periods (P = 0.01), and increased mean CAP rate/non-rapid eye movement (NREM) (P <0.001). Only four PWS patients had apnea/hypopnea index (AHI) ≥ 10. Conversely, significant nocturnal oxygen desaturation was frequent (83% of patients) and independent from apneas or hypopneas. In the PWS group, CAP rate/NREM showed a significant negative correlation with MSLT score (P = 0.02) independently from arousals, respiratory disturbance variables, severity of illness measured by Holm's score or body mass index (BMI). PWS patients with CAP expression characterized by higher proportion of A1 subtypes presented less severe GH deficiency (P = 0.01). Conclusions: Our study suggests a relationship between hypersomnia and CAP rate, and between CAP expression and GH secretory pattern in PWS, possibly reflecting underlying central dysfunctions.

Original languageEnglish
Pages (from-to)627-633
Number of pages7
JournalSleep Medicine
Volume7
Issue number8
DOIs
Publication statusPublished - Dec 2006

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Disorders of Excessive Somnolence
Prader-Willi Syndrome
Growth Hormone
Sleep
REM Sleep
Apnea
Eye Movements
Respiration
Growth Hormone-Releasing Hormone
Polysomnography
Sleep Apnea Syndromes
Arousal
Arginine
Young Adult
Healthy Volunteers
Body Mass Index

Keywords

  • Apnea/hypopnea index
  • Cycling alternating pattern
  • GH deficiency
  • Hypersomnia
  • Multiple sleep latency test
  • Prader-Willi syndrome

ASJC Scopus subject areas

  • Dentistry(all)
  • Ophthalmology
  • Pulmonary and Respiratory Medicine
  • Neurology

Cite this

Sleep cycling alternating pattern (CAP) expression is associated with hypersomnia and GH secretory pattern in Prader-Willi syndrome. / Priano, Lorenzo; Grugni, Graziano; Miscio, Giacinta; Guastamacchia, Giulia; Toffolet, Lorenzo; Sartorio, Alessandro; Mauro, Alessandro.

In: Sleep Medicine, Vol. 7, No. 8, 12.2006, p. 627-633.

Research output: Contribution to journalArticle

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abstract = "Background and purpose: Hypersomnia, sleep-disordered breathing and narcoleptic traits such as rapid eye movement (REM) sleep onset periods (SOREMPs) have been reported in Prader-Willi syndrome (PWS). In a group of young adult patients with genetically confirmed PWS we evaluated sleep and breathing polysomnographically, including cycling alternating pattern (CAP), and we analyzed the potential interacting role of sleep variables, sleep-related breathing abnormalities, hypersomnia, severity of illness variables and growth hormone (GH) secretory pattern. Patients and methods: Eleven males and 7 females (mean age: 27.5±5.5 years) were submitted to a full night of complete polysomnography and the multiple sleep latency test (MSLT). GH secretory pattern was evaluated by a standard GH-releasing hormone plus arginine test. Sixteen non-obese healthy subjects without sleep disturbances were recruited as controls. Results: Compared to controls PWS patients showed reduced mean MSLT score (P <0.001), reduced mean latency of sleep (P = 0.03), increased REM sleep periods (P = 0.01), and increased mean CAP rate/non-rapid eye movement (NREM) (P <0.001). Only four PWS patients had apnea/hypopnea index (AHI) ≥ 10. Conversely, significant nocturnal oxygen desaturation was frequent (83{\%} of patients) and independent from apneas or hypopneas. In the PWS group, CAP rate/NREM showed a significant negative correlation with MSLT score (P = 0.02) independently from arousals, respiratory disturbance variables, severity of illness measured by Holm's score or body mass index (BMI). PWS patients with CAP expression characterized by higher proportion of A1 subtypes presented less severe GH deficiency (P = 0.01). Conclusions: Our study suggests a relationship between hypersomnia and CAP rate, and between CAP expression and GH secretory pattern in PWS, possibly reflecting underlying central dysfunctions.",
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T1 - Sleep cycling alternating pattern (CAP) expression is associated with hypersomnia and GH secretory pattern in Prader-Willi syndrome

AU - Priano, Lorenzo

AU - Grugni, Graziano

AU - Miscio, Giacinta

AU - Guastamacchia, Giulia

AU - Toffolet, Lorenzo

AU - Sartorio, Alessandro

AU - Mauro, Alessandro

PY - 2006/12

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AB - Background and purpose: Hypersomnia, sleep-disordered breathing and narcoleptic traits such as rapid eye movement (REM) sleep onset periods (SOREMPs) have been reported in Prader-Willi syndrome (PWS). In a group of young adult patients with genetically confirmed PWS we evaluated sleep and breathing polysomnographically, including cycling alternating pattern (CAP), and we analyzed the potential interacting role of sleep variables, sleep-related breathing abnormalities, hypersomnia, severity of illness variables and growth hormone (GH) secretory pattern. Patients and methods: Eleven males and 7 females (mean age: 27.5±5.5 years) were submitted to a full night of complete polysomnography and the multiple sleep latency test (MSLT). GH secretory pattern was evaluated by a standard GH-releasing hormone plus arginine test. Sixteen non-obese healthy subjects without sleep disturbances were recruited as controls. Results: Compared to controls PWS patients showed reduced mean MSLT score (P <0.001), reduced mean latency of sleep (P = 0.03), increased REM sleep periods (P = 0.01), and increased mean CAP rate/non-rapid eye movement (NREM) (P <0.001). Only four PWS patients had apnea/hypopnea index (AHI) ≥ 10. Conversely, significant nocturnal oxygen desaturation was frequent (83% of patients) and independent from apneas or hypopneas. In the PWS group, CAP rate/NREM showed a significant negative correlation with MSLT score (P = 0.02) independently from arousals, respiratory disturbance variables, severity of illness measured by Holm's score or body mass index (BMI). PWS patients with CAP expression characterized by higher proportion of A1 subtypes presented less severe GH deficiency (P = 0.01). Conclusions: Our study suggests a relationship between hypersomnia and CAP rate, and between CAP expression and GH secretory pattern in PWS, possibly reflecting underlying central dysfunctions.

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KW - GH deficiency

KW - Hypersomnia

KW - Multiple sleep latency test

KW - Prader-Willi syndrome

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