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Effects of Growth Hormone Treatment on Sleep-Related Parameters in Adults With Prader-Willi Syndrome

CONTEXT: Prader-Willi syndrome (PWS) is a rare, genetic, multisymptom, neurodevelopmental disease due to lack of the expression of the paternal genes in the q11 to q13 region of chromosome 15. The main characteristics of PWS are muscular hypotonia, hyperphagia, obesity, behavioral problems, cognitiv...

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Autores principales: Shukur, Hasanain Hamid, Hussain-Alkhateeb, Laith, Farholt, Stense, Nørregaard, Ole, Jørgensen, Anders Palmstrøm, Hoybye, Charlotte
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8372636/
https://www.ncbi.nlm.nih.gov/pubmed/33950234
http://dx.doi.org/10.1210/clinem/dgab300
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author Shukur, Hasanain Hamid
Hussain-Alkhateeb, Laith
Farholt, Stense
Nørregaard, Ole
Jørgensen, Anders Palmstrøm
Hoybye, Charlotte
author_facet Shukur, Hasanain Hamid
Hussain-Alkhateeb, Laith
Farholt, Stense
Nørregaard, Ole
Jørgensen, Anders Palmstrøm
Hoybye, Charlotte
author_sort Shukur, Hasanain Hamid
collection PubMed
description CONTEXT: Prader-Willi syndrome (PWS) is a rare, genetic, multisymptom, neurodevelopmental disease due to lack of the expression of the paternal genes in the q11 to q13 region of chromosome 15. The main characteristics of PWS are muscular hypotonia, hyperphagia, obesity, behavioral problems, cognitive disabilities, and endocrine deficiencies, including growth hormone (GH) deficiency. Sleep apnea and abnormal sleep patterns are common in PWS. GH treatment might theoretically have a negative impact on respiration. OBJECTIVE: Here we present the effect of GH treatment on polysomnographic measurements. METHODS: Thirty-seven adults, 15 men and 22 women, with confirmed PWS were randomly assigned to 1 year of GH treatment (n = 19) or placebo (n = 18) followed by 2 years of GH treatment to all. Polysomnographic measurements were performed every 6 months. A mixed-effect regression model was used for comparison over time in the subgroup that received GH for 3 years. RESULTS: At baseline median age was 29.5 years, body mass index 27.1, insulin-like growth factor 115 µg/L, apnea-hypopnea index (AHI) 1.4 (range, 0.0-13.9), and sleep efficiency (SE) 89.0% (range, 41.0%-99.0%). No differences in sleep or respiratory parameters were seen between GH- and placebo-treated patients. SE continuously improved throughout the study, also after adjustment for BMI, and the length of the longest apnea increased. AHI inconsistently increased within normal range. CONCLUSION: SE improved during GH treatment and no clinical, significantly negative impact on respiration was seen. The etiology of breathing disorders is multifactorial and awareness of them should always be present in adults with PWS with or without GH treatment.
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spelling pubmed-83726362021-08-20 Effects of Growth Hormone Treatment on Sleep-Related Parameters in Adults With Prader-Willi Syndrome Shukur, Hasanain Hamid Hussain-Alkhateeb, Laith Farholt, Stense Nørregaard, Ole Jørgensen, Anders Palmstrøm Hoybye, Charlotte J Clin Endocrinol Metab Online Only Articles CONTEXT: Prader-Willi syndrome (PWS) is a rare, genetic, multisymptom, neurodevelopmental disease due to lack of the expression of the paternal genes in the q11 to q13 region of chromosome 15. The main characteristics of PWS are muscular hypotonia, hyperphagia, obesity, behavioral problems, cognitive disabilities, and endocrine deficiencies, including growth hormone (GH) deficiency. Sleep apnea and abnormal sleep patterns are common in PWS. GH treatment might theoretically have a negative impact on respiration. OBJECTIVE: Here we present the effect of GH treatment on polysomnographic measurements. METHODS: Thirty-seven adults, 15 men and 22 women, with confirmed PWS were randomly assigned to 1 year of GH treatment (n = 19) or placebo (n = 18) followed by 2 years of GH treatment to all. Polysomnographic measurements were performed every 6 months. A mixed-effect regression model was used for comparison over time in the subgroup that received GH for 3 years. RESULTS: At baseline median age was 29.5 years, body mass index 27.1, insulin-like growth factor 115 µg/L, apnea-hypopnea index (AHI) 1.4 (range, 0.0-13.9), and sleep efficiency (SE) 89.0% (range, 41.0%-99.0%). No differences in sleep or respiratory parameters were seen between GH- and placebo-treated patients. SE continuously improved throughout the study, also after adjustment for BMI, and the length of the longest apnea increased. AHI inconsistently increased within normal range. CONCLUSION: SE improved during GH treatment and no clinical, significantly negative impact on respiration was seen. The etiology of breathing disorders is multifactorial and awareness of them should always be present in adults with PWS with or without GH treatment. Oxford University Press 2021-05-05 /pmc/articles/PMC8372636/ /pubmed/33950234 http://dx.doi.org/10.1210/clinem/dgab300 Text en © The Author(s) 2021. Published by Oxford University Press on behalf of the Endocrine Society. https://creativecommons.org/licenses/by-nc-nd/4.0/This is an Open Access article distributed under the terms of the Creative Commons Attribution-NonCommercial-NoDerivs licence (http://creativecommons.org/licenses/by-nc-nd/4.0/ (https://creativecommons.org/licenses/by-nc-nd/4.0/) ), which permits non-commercial reproduction and distribution of the work, in any medium, provided the original work is not altered or transformed in any way, and that the work is properly cited. For commercial re-use, please contact journals.permissions@oup.com
spellingShingle Online Only Articles
Shukur, Hasanain Hamid
Hussain-Alkhateeb, Laith
Farholt, Stense
Nørregaard, Ole
Jørgensen, Anders Palmstrøm
Hoybye, Charlotte
Effects of Growth Hormone Treatment on Sleep-Related Parameters in Adults With Prader-Willi Syndrome
title Effects of Growth Hormone Treatment on Sleep-Related Parameters in Adults With Prader-Willi Syndrome
title_full Effects of Growth Hormone Treatment on Sleep-Related Parameters in Adults With Prader-Willi Syndrome
title_fullStr Effects of Growth Hormone Treatment on Sleep-Related Parameters in Adults With Prader-Willi Syndrome
title_full_unstemmed Effects of Growth Hormone Treatment on Sleep-Related Parameters in Adults With Prader-Willi Syndrome
title_short Effects of Growth Hormone Treatment on Sleep-Related Parameters in Adults With Prader-Willi Syndrome
title_sort effects of growth hormone treatment on sleep-related parameters in adults with prader-willi syndrome
topic Online Only Articles
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8372636/
https://www.ncbi.nlm.nih.gov/pubmed/33950234
http://dx.doi.org/10.1210/clinem/dgab300
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