Literature DB >> 29192378

Comparing Parental Well-Being and Its Determinants Across Three Different Genetic Disorders Causing Intellectual Disability.

Yuka Mori1,2, Jenny Downs1,2,3, Kingsley Wong1,2, Jane Heyworth4, Helen Leonard5,6.   

Abstract

Using the Short Form 12 Health Survey this cross-sectional study examined parental well-being in caregivers of children with one of three genetic disorders associated with intellectual disability; Down syndrome, Rett syndrome and the CDKL5 disorder. Data were sourced from the Western Australian Down Syndrome (n = 291), Australian Rett Syndrome (n = 187) and International CDKL5 Disorder (n = 168) Databases. Among 596 mothers (median age, years 43.7; 24.6-72.2), emotional well-being was poorer than general female populations across age groups. Multivariate linear regression identified the poorest well-being in parents of children with the CDKL5 disorder, a rare but severe and complex encephalopathy, and negative associations with increased clinical severity irrespective of diagnosis. These findings are important for those providing healthcare and social services for these populations.

Entities:  

Keywords:  Down syndrome; Genetic disorder; Intellectual disability; Parental well-being; Rett syndrome; SF-12

Mesh:

Year:  2018        PMID: 29192378     DOI: 10.1007/s10803-017-3420-x

Source DB:  PubMed          Journal:  J Autism Dev Disord        ISSN: 0162-3257


  56 in total

1.  The phenotype associated with a large deletion on MECP2.

Authors:  Ami Bebbington; Jenny Downs; Alan Percy; Mercé Pineda; Bruria Ben Zeev; Nadia Bahi-Buisson; Helen Leonard
Journal:  Eur J Hum Genet       Date:  2012-04-04       Impact factor: 4.246

2.  The association between behavior and genotype in Rett syndrome using the Australian Rett Syndrome Database.

Authors:  Laila Robertson; Sonĵa E Hall; Peter Jacoby; Carolyn Ellaway; Nick de Klerk; Helen Leonard
Journal:  Am J Med Genet B Neuropsychiatr Genet       Date:  2006-03-05       Impact factor: 3.568

3.  The CDKL5 disorder is an independent clinical entity associated with early-onset encephalopathy.

Authors:  Stephanie Fehr; Meredith Wilson; Jenny Downs; Simon Williams; Alessandra Murgia; Stefano Sartori; Marilena Vecchi; Gladys Ho; Roberta Polli; Stavroula Psoni; Xinhua Bao; Nick de Klerk; Helen Leonard; John Christodoulou
Journal:  Eur J Hum Genet       Date:  2012-08-08       Impact factor: 4.246

4.  Using online health communication to manage chronic sorrow: mothers of children with rare diseases speak.

Authors:  Adriana D Glenn
Journal:  J Pediatr Nurs       Date:  2014-10-05       Impact factor: 2.145

5.  How families of children with complex care needs participate in everyday life.

Authors:  Roberta Lynn Woodgate; Marie Edwards; Jacquie Ripat
Journal:  Soc Sci Med       Date:  2012-08-10       Impact factor: 4.634

6.  Genome sequencing identifies major causes of severe intellectual disability.

Authors:  Christian Gilissen; Jayne Y Hehir-Kwa; Djie Tjwan Thung; Maartje van de Vorst; Bregje W M van Bon; Marjolein H Willemsen; Michael Kwint; Irene M Janssen; Alexander Hoischen; Annette Schenck; Richard Leach; Robert Klein; Rick Tearle; Tan Bo; Rolph Pfundt; Helger G Yntema; Bert B A de Vries; Tjitske Kleefstra; Han G Brunner; Lisenka E L M Vissers; Joris A Veltman
Journal:  Nature       Date:  2014-06-04       Impact factor: 49.962

7.  Social support and mastery influence the association between stress and poor physical health in parents caring for children with developmental disabilities.

Authors:  Joanne Cantwell; Orla T Muldoon; Stephen Gallagher
Journal:  Res Dev Disabil       Date:  2014-06-11

8.  Obtaining a genetic diagnosis in a child with disability: impact on parental quality of life.

Authors:  M Lingen; L Albers; M Borchers; S Haass; J Gärtner; S Schröder; L Goldbeck; R von Kries; K Brockmann; B Zirn
Journal:  Clin Genet       Date:  2015-07-14       Impact factor: 4.438

9.  Extracting more information from behaviour checklists by using components of mean based scores.

Authors:  John R Taffe; Bruce J Tonge; Kylie M Gray; Stewart L Einfeld
Journal:  Int J Methods Psychiatr Res       Date:  2008       Impact factor: 4.035

10.  Twenty years of surveillance in Rett syndrome: what does this tell us?

Authors:  Alison Anderson; Kingsley Wong; Peter Jacoby; Jenny Downs; Helen Leonard
Journal:  Orphanet J Rare Dis       Date:  2014-06-19       Impact factor: 4.123

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  5 in total

1.  The effect of functioning on Quality of Life Inventory-Disability measured quality of life is not mediated or moderated by parental psychological distress.

Authors:  A J O Whitehouse; P Jacoby; D Reddihough; H Leonard; K Williams; Jenny Downs
Journal:  Qual Life Res       Date:  2021-05-03       Impact factor: 4.147

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

Authors:  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
Journal:  J Autism Dev Disord       Date:  2021-09-08

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

Authors:  Jacqueline Fitzgerald; Louise Gallagher
Journal:  J Intellect Disabil       Date:  2021-04-19

Review 4.  Improving clinical trial readiness to accelerate development of new therapeutics for Rett syndrome.

Authors:  Helen Leonard; Wendy Gold; Rodney Samaco; Mustafa Sahin; Timothy Benke; Jenny Downs
Journal:  Orphanet J Rare Dis       Date:  2022-03-04       Impact factor: 4.123

5.  The Family-Centred Practices Scale: Psychometric properties of the Spanish version for use with families with children with Down syndrome receiving early childhood intervention.

Authors:  María Auxiliadora Robles-Bello; David Sánchez-Teruel
Journal:  Child Care Health Dev       Date:  2022-01-30       Impact factor: 2.943

  5 in total

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