Life Satisfaction Among Mothers of Individuals with Prader-Willi Syndrome

Shivers, Carolyn M.; Leonczyk, Caroline; Dykens, Elisabeth M.

doi:10.1007/s10803-016-2741-5

Cited by 17 publications

(16 citation statements)

References 59 publications

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“…A number of studies indicated that child age was not associated with parental stress ( Byiers et al, 2014 ; Fidler et al, 2000 ; Sarimski, 2010 ; Wulffaert et al, 2010 ), life satisfaction ( Shivers et al, 2016 ), strain ( Jacob et al., 2017 ; Luescher et al., 1999 ) or quality of life ( Laurvick et al, 2006 ) in a number of rare genetic syndromes. However, other studies demonstrated an association between increased child age and greater parent stress ( Farmer et al, 2006 ; Hodapp et al, 1997 ; Sarimski, 1997b ; Wulffaert et al, 2009 ).…”

Section: Resultsmentioning

confidence: 99%

Parental stress and adjustment in the context of rare genetic syndromes: A scoping review

Fitzgerald

Gallagher

2021

J Intellect Disabil

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Chromosomal abnormalities are now considered a common cause of intellectual disability. With increased genetic testing, phenotyping and technological advancements, many new syndromes have been identified. This review sought to explore parental stress and adjustment in the context of rare genetic syndromes to evaluate their clinical impact. A systematic review of English peer-reviewed literature across three databases (PsycINFO, Medline, CINAHL) was completed and 69 articles were included. Parents of children with rare genetic syndromes experienced greater distress relative to other disabilities. Differences in parental wellbeing were syndrome-specific relative to ASD thus demonstrating the need to consider the contribution of syndrome-specific phenotypes. Child emotional and behavioural difficulties were the most consistent predictor of parental distress. Research reflecting other factors such as physical health, syndrome-specific behaviours, benefit finding and, parental appraisal in the context of a rare genetic aetiology is required in order to support parental adjustment in these conditions.

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Section: Resultsmentioning

confidence: 99%

Parental stress and adjustment in the context of rare genetic syndromes: A scoping review

Fitzgerald

Gallagher

2021

J Intellect Disabil

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“…Caregiver demands in PWS also include intensive supervision due to the need to limit access to food . Excessive food‐seeking behaviours have been negatively correlated with maternal life satisfaction ( n = 123; r = −0.25, P = 0.001) and are associated with higher levels of family stress . Greater availability of food outside the home environment and difficulties in accessing timely respite with carers who have an adequate understanding of PWS may also restrict social activities .…”

Section: Resultsmentioning

confidence: 99%

Requirements for improving health and well‐being of children with Prader‐Willi syndrome and their families

Mackay

McCallum

Ambler

et al. 2019

J Paediatrics Child Health

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Prader‐Willi syndrome (PWS) is a rare genetic condition with multi‐system involvement. The literature was reviewed to describe neurodevelopment and the behavioural phenotype, endocrine and metabolic disorders and respiratory and sleep functioning. Implications for child and family quality of life were explored. Challenging behaviours contribute to poorer well‐being and quality of life for both the child and caregiver. Recent evidence indicates healthy outcomes of weight and height can be achieved with growth hormone therapy and dietary restriction and should be the current target for all individuals with PWS. Gaps in the literature included therapies to manage challenging behaviours, as well as understanding the effects of growth hormone on respiratory and sleep function. New knowledge regarding the transition of children and families from schooling and paediatric health services to employment, accommodation and adult health services is also needed. Developing a national population‐based registry could address these knowledge gaps and inform advocacy for support services that improve the well‐being of individuals with PWS and their families.

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“…The behavioural phenotypebehaviour shown more commonly in individuals with PWS than in those without (Dykens 1995) includes strong resistance to change and frequent temper outbursts, a substantial proportion of which follow changes to routines or expectations (Holland et al 2003;Moss et al 2009;Woodcock et al 2009a;Tunnicliffe et al 2014). Temper outbursts represent one of the most impairing aspects of PWS (Manning et al 2016;Shivers et al 2016).…”

Section: Resultsmentioning

confidence: 99%

Rigidity in routines and the development of resistance to change in individuals with Prader–Willi syndrome

Haig

Woodcock

2017

J intellect Disabil Res

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Because task switching appears to evidence a period of high developmental sensitivity during early primary school years, we propose that this period may represent a critical time when increasing flexibility in the routines of children with PWS could limit the development of resistance to change. However, a careful balance would need to be struck, given the apparent benefit of rigid routines on current behaviour. Further work in this area is much needed.

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Life Satisfaction Among Mothers of Individuals with Prader-Willi Syndrome

Cited by 17 publications

References 59 publications

Parental stress and adjustment in the context of rare genetic syndromes: A scoping review

Parental stress and adjustment in the context of rare genetic syndromes: A scoping review

Requirements for improving health and well‐being of children with Prader‐Willi syndrome and their families

Rigidity in routines and the development of resistance to change in individuals with Prader–Willi syndrome

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