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Sleep‐disordered breathing in Australian children with Prader‐Willi syndrome following initiation of growth hormone therapy

AIM: In children with Prader‐Willi syndrome (PWS), growth hormone (GH) improves height and body composition; however, may be associated with worsening sleep‐disordered breathing (SDB). Some studies have reported less SDB after GH initiation, but follow‐up with polysomnography is still advised in mos...

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Autores principales: Caudri, Daan, Nixon, Gillian M, Nielsen, Aleisha, Mai, Linda, Hafekost, Claire R, Kapur, Nitin, Seton, Chris, Tai, Andrew, Blecher, Greg, Ambler, Geoff, Bergman, Philip B, Vora, Komal A, Crock, Patricia, Verge, Charles F, Tham, Elaine, Musthaffa, Yassmin, Lafferty, Antony R, Jacoby, Peter, Wilson, Andrew C, Downs, Jenny, Choong, Catherine S
Formato: Online Artículo Texto
Lenguaje:English
Publicado: John Wiley & Sons Australia, Ltd. 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9290886/
https://www.ncbi.nlm.nih.gov/pubmed/34397126
http://dx.doi.org/10.1111/jpc.15691
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author Caudri, Daan
Nixon, Gillian M
Nielsen, Aleisha
Mai, Linda
Hafekost, Claire R
Kapur, Nitin
Seton, Chris
Tai, Andrew
Blecher, Greg
Ambler, Geoff
Bergman, Philip B
Vora, Komal A
Crock, Patricia
Verge, Charles F
Tham, Elaine
Musthaffa, Yassmin
Lafferty, Antony R
Jacoby, Peter
Wilson, Andrew C
Downs, Jenny
Choong, Catherine S
author_facet Caudri, Daan
Nixon, Gillian M
Nielsen, Aleisha
Mai, Linda
Hafekost, Claire R
Kapur, Nitin
Seton, Chris
Tai, Andrew
Blecher, Greg
Ambler, Geoff
Bergman, Philip B
Vora, Komal A
Crock, Patricia
Verge, Charles F
Tham, Elaine
Musthaffa, Yassmin
Lafferty, Antony R
Jacoby, Peter
Wilson, Andrew C
Downs, Jenny
Choong, Catherine S
author_sort Caudri, Daan
collection PubMed
description AIM: In children with Prader‐Willi syndrome (PWS), growth hormone (GH) improves height and body composition; however, may be associated with worsening sleep‐disordered breathing (SDB). Some studies have reported less SDB after GH initiation, but follow‐up with polysomnography is still advised in most clinical guidelines. METHODS: This retrospective, multicentre study, included children with PWS treated with GH at seven PWS treatment centres in Australia over the last 18 years. A paired analysis comparing polysomnographic measures of central and obstructive SDB in the same child, before and after GH initiation was performed with Wilcoxon signed‐rank test. The proportion of children who developed moderate/severe obstructive sleep apnoea (OSA) was calculated with their binomial confidence intervals. RESULTS: We included 112 patients with available paired data. The median age at start of GH was 1.9 years (range 0.1–13.5 years). Median obstructive apnoea hypopnoea index (AHI) at baseline was 0.43/h (range 0–32.9); 35% had an obstructive AHI above 1.0/h. Follow‐up polysomnography within 2 years after the start of GH was available in 94 children who did not receive OSA treatment. After GH initiation, there was no change in central AHI. The median obstructive AHI did not increase significantly (P = 0.13), but 12 children (13%, CI(95%) 7–21%) developed moderate/severe OSA, with clinical management implications. CONCLUSIONS: Our findings of a worsening of OSA severity in 13% of children with PWS support current advice to perform polysomnography after GH initiation. Early identification of worsening OSA may prevent severe sequelae in a subgroup of children.
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spelling pubmed-92908862022-07-20 Sleep‐disordered breathing in Australian children with Prader‐Willi syndrome following initiation of growth hormone therapy Caudri, Daan Nixon, Gillian M Nielsen, Aleisha Mai, Linda Hafekost, Claire R Kapur, Nitin Seton, Chris Tai, Andrew Blecher, Greg Ambler, Geoff Bergman, Philip B Vora, Komal A Crock, Patricia Verge, Charles F Tham, Elaine Musthaffa, Yassmin Lafferty, Antony R Jacoby, Peter Wilson, Andrew C Downs, Jenny Choong, Catherine S J Paediatr Child Health Original Articles AIM: In children with Prader‐Willi syndrome (PWS), growth hormone (GH) improves height and body composition; however, may be associated with worsening sleep‐disordered breathing (SDB). Some studies have reported less SDB after GH initiation, but follow‐up with polysomnography is still advised in most clinical guidelines. METHODS: This retrospective, multicentre study, included children with PWS treated with GH at seven PWS treatment centres in Australia over the last 18 years. A paired analysis comparing polysomnographic measures of central and obstructive SDB in the same child, before and after GH initiation was performed with Wilcoxon signed‐rank test. The proportion of children who developed moderate/severe obstructive sleep apnoea (OSA) was calculated with their binomial confidence intervals. RESULTS: We included 112 patients with available paired data. The median age at start of GH was 1.9 years (range 0.1–13.5 years). Median obstructive apnoea hypopnoea index (AHI) at baseline was 0.43/h (range 0–32.9); 35% had an obstructive AHI above 1.0/h. Follow‐up polysomnography within 2 years after the start of GH was available in 94 children who did not receive OSA treatment. After GH initiation, there was no change in central AHI. The median obstructive AHI did not increase significantly (P = 0.13), but 12 children (13%, CI(95%) 7–21%) developed moderate/severe OSA, with clinical management implications. CONCLUSIONS: Our findings of a worsening of OSA severity in 13% of children with PWS support current advice to perform polysomnography after GH initiation. Early identification of worsening OSA may prevent severe sequelae in a subgroup of children. John Wiley & Sons Australia, Ltd. 2021-08-16 2022-02 /pmc/articles/PMC9290886/ /pubmed/34397126 http://dx.doi.org/10.1111/jpc.15691 Text en © 2021 The Authors. Journal of Paediatrics and Child Health published by John Wiley & Sons Australia, Ltd on behalf of Paediatrics and Child Health Division (The Royal Australasian College of Physicians). https://creativecommons.org/licenses/by-nc-nd/4.0/This is an open access article under the terms of the http://creativecommons.org/licenses/by-nc-nd/4.0/ (https://creativecommons.org/licenses/by-nc-nd/4.0/) License, which permits use and distribution in any medium, provided the original work is properly cited, the use is non‐commercial and no modifications or adaptations are made.
spellingShingle Original Articles
Caudri, Daan
Nixon, Gillian M
Nielsen, Aleisha
Mai, Linda
Hafekost, Claire R
Kapur, Nitin
Seton, Chris
Tai, Andrew
Blecher, Greg
Ambler, Geoff
Bergman, Philip B
Vora, Komal A
Crock, Patricia
Verge, Charles F
Tham, Elaine
Musthaffa, Yassmin
Lafferty, Antony R
Jacoby, Peter
Wilson, Andrew C
Downs, Jenny
Choong, Catherine S
Sleep‐disordered breathing in Australian children with Prader‐Willi syndrome following initiation of growth hormone therapy
title Sleep‐disordered breathing in Australian children with Prader‐Willi syndrome following initiation of growth hormone therapy
title_full Sleep‐disordered breathing in Australian children with Prader‐Willi syndrome following initiation of growth hormone therapy
title_fullStr Sleep‐disordered breathing in Australian children with Prader‐Willi syndrome following initiation of growth hormone therapy
title_full_unstemmed Sleep‐disordered breathing in Australian children with Prader‐Willi syndrome following initiation of growth hormone therapy
title_short Sleep‐disordered breathing in Australian children with Prader‐Willi syndrome following initiation of growth hormone therapy
title_sort sleep‐disordered breathing in australian children with prader‐willi syndrome following initiation of growth hormone therapy
topic Original Articles
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9290886/
https://www.ncbi.nlm.nih.gov/pubmed/34397126
http://dx.doi.org/10.1111/jpc.15691
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