Associations Between Hyperphagia, Symptoms of Sleep Breathing Disorder, Behaviour Difficulties and Caregiver Well-Being in Prader-Willi Syndrome: A Preliminary Study

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

doi:10.1007/s10803-021-05265-5

Associations Between Hyperphagia, Symptoms of Sleep Breathing Disorder, Behaviour Difficulties and Caregiver Well-Being in Prader-Willi Syndrome: A Preliminary Study

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. ChoongJenny Downs^*

^*Corresponding author for this work

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3 Citations (Scopus)

Abstract

Prader-Willi syndrome (PWS) is a rare genetic disorder characterised by neurodevelopmental delays, hyperphagia, difficulties with social communication and challenging behaviours. Individuals require intensive supervision from caregivers which may negatively affect caregiver quality of life. This study used data collected in the Australasian PWS Registry (n = 50, mean age 11.2 years) to evaluate associations between child behaviours and caregiver mental well-being. Symptoms of sleep-related breathing disorder, child depression and social difficulties were associated with poorer caregiver mental and physical well-being. Growth hormone therapy use was associated with better caregiver mental and physical well-being. Optimising management of problematic behaviours and sleep disturbances have the potential to support caregivers who are the most vital network of support for individuals affected by PWS.

Original language	English
Pages (from-to)	3877-3889
Number of pages	13
Journal	Journal of Autism and Developmental Disorders
Volume	52
Issue number	9
DOIs	https://doi.org/10.1007/s10803-021-05265-5
Publication status	Published - Sept 2022

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Mackay, J., Nixon, G. M., Lafferty, A. R., Ambler, G., Kapur, N., Bergman, P. B., Schofield, C., Seton, C., Tai, A., Tham, E., Vora, K., Crock, P., Verge, C., Musthaffa, Y., Blecher, G., Caudri, D., Leonard, H., Jacoby, P., Wilson, A., ... Downs, J. (2022). Associations Between Hyperphagia, Symptoms of Sleep Breathing Disorder, Behaviour Difficulties and Caregiver Well-Being in Prader-Willi Syndrome: A Preliminary Study. Journal of Autism and Developmental Disorders, 52(9), 3877-3889. https://doi.org/10.1007/s10803-021-05265-5

@article{182b95f3bbfd4f23bb4dc98bf52177af,

title = "Associations Between Hyperphagia, Symptoms of Sleep Breathing Disorder, Behaviour Difficulties and Caregiver Well-Being in Prader-Willi Syndrome: A Preliminary Study",

abstract = "Prader-Willi syndrome (PWS) is a rare genetic disorder characterised by neurodevelopmental delays, hyperphagia, difficulties with social communication and challenging behaviours. Individuals require intensive supervision from caregivers which may negatively affect caregiver quality of life. This study used data collected in the Australasian PWS Registry (n = 50, mean age 11.2 years) to evaluate associations between child behaviours and caregiver mental well-being. Symptoms of sleep-related breathing disorder, child depression and social difficulties were associated with poorer caregiver mental and physical well-being. Growth hormone therapy use was associated with better caregiver mental and physical well-being. Optimising management of problematic behaviours and sleep disturbances have the potential to support caregivers who are the most vital network of support for individuals affected by PWS.",

keywords = "Growth hormone, Hyperphagia; behaviour, Parental well-being, Prader-Willi syndrome, Sleep",

author = "Jessica Mackay and Nixon, \{Gillian M.\} and Lafferty, \{Antony R.\} and Geoff Ambler and Nitin Kapur and Bergman, \{Philip B.\} and Cara Schofield and Chris Seton and Andrew Tai and Elaine Tham and Komal Vora and Patricia Crock and Charles Verge and Yassmin Musthaffa and Greg Blecher and Daan Caudri and Helen Leonard and Peter Jacoby and Andrew Wilson and Choong, \{Catherine S.\} and Jenny Downs",

note = "Publisher Copyright: {\textcopyright} 2021, The Author(s), under exclusive licence to Springer Science+Business Media, LLC, part of Springer Nature.",

year = "2022",

month = sep,

doi = "10.1007/s10803-021-05265-5",

language = "English",

volume = "52",

pages = "3877--3889",

journal = "Journal of Autism and Developmental Disorders",

issn = "0162-3257",

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Mackay, J, Nixon, GM, Lafferty, AR, Ambler, G, Kapur, N, Bergman, PB, Schofield, C, Seton, C, Tai, A, Tham, E, Vora, K, Crock, P, Verge, C, Musthaffa, Y, Blecher, G, Caudri, D, Leonard, H, Jacoby, P, Wilson, A, Choong, CS & Downs, J 2022, 'Associations Between Hyperphagia, Symptoms of Sleep Breathing Disorder, Behaviour Difficulties and Caregiver Well-Being in Prader-Willi Syndrome: A Preliminary Study', Journal of Autism and Developmental Disorders, vol. 52, no. 9, pp. 3877-3889. https://doi.org/10.1007/s10803-021-05265-5

Associations Between Hyperphagia, Symptoms of Sleep Breathing Disorder, Behaviour Difficulties and Caregiver Well-Being in Prader-Willi Syndrome: A Preliminary Study. / Mackay, Jessica; Nixon, Gillian M.; Lafferty, Antony R. et al.
In: Journal of Autism and Developmental Disorders, Vol. 52, No. 9, 09.2022, p. 3877-3889.

Research output: Contribution to journal › Article › peer-review

TY - JOUR

T1 - Associations Between Hyperphagia, Symptoms of Sleep Breathing Disorder, Behaviour Difficulties and Caregiver Well-Being in Prader-Willi Syndrome

T2 - A Preliminary Study

AU - Mackay, Jessica

AU - Nixon, Gillian M.

AU - Lafferty, Antony R.

AU - Ambler, Geoff

AU - Kapur, Nitin

AU - Bergman, Philip B.

AU - Schofield, Cara

AU - Seton, Chris

AU - Tai, Andrew

AU - Tham, Elaine

AU - Vora, Komal

AU - Crock, Patricia

AU - Verge, Charles

AU - Musthaffa, Yassmin

AU - Blecher, Greg

AU - Caudri, Daan

AU - Leonard, Helen

AU - Jacoby, Peter

AU - Wilson, Andrew

AU - Choong, Catherine S.

AU - Downs, Jenny

PY - 2022/9

Y1 - 2022/9

N2 - Prader-Willi syndrome (PWS) is a rare genetic disorder characterised by neurodevelopmental delays, hyperphagia, difficulties with social communication and challenging behaviours. Individuals require intensive supervision from caregivers which may negatively affect caregiver quality of life. This study used data collected in the Australasian PWS Registry (n = 50, mean age 11.2 years) to evaluate associations between child behaviours and caregiver mental well-being. Symptoms of sleep-related breathing disorder, child depression and social difficulties were associated with poorer caregiver mental and physical well-being. Growth hormone therapy use was associated with better caregiver mental and physical well-being. Optimising management of problematic behaviours and sleep disturbances have the potential to support caregivers who are the most vital network of support for individuals affected by PWS.

AB - Prader-Willi syndrome (PWS) is a rare genetic disorder characterised by neurodevelopmental delays, hyperphagia, difficulties with social communication and challenging behaviours. Individuals require intensive supervision from caregivers which may negatively affect caregiver quality of life. This study used data collected in the Australasian PWS Registry (n = 50, mean age 11.2 years) to evaluate associations between child behaviours and caregiver mental well-being. Symptoms of sleep-related breathing disorder, child depression and social difficulties were associated with poorer caregiver mental and physical well-being. Growth hormone therapy use was associated with better caregiver mental and physical well-being. Optimising management of problematic behaviours and sleep disturbances have the potential to support caregivers who are the most vital network of support for individuals affected by PWS.

KW - Growth hormone

KW - Hyperphagia; behaviour

KW - Parental well-being

KW - Prader-Willi syndrome

KW - Sleep

UR - https://www.scopus.com/pages/publications/85114432689

U2 - 10.1007/s10803-021-05265-5

DO - 10.1007/s10803-021-05265-5

M3 - Article

SN - 0162-3257

VL - 52

SP - 3877

EP - 3889

JO - Journal of Autism and Developmental Disorders

JF - Journal of Autism and Developmental Disorders

IS - 9

ER -

Associations Between Hyperphagia, Symptoms of Sleep Breathing Disorder, Behaviour Difficulties and Caregiver Well-Being in Prader-Willi Syndrome: A Preliminary Study

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