Comparing Parental Well-Being and Its Determinants Across Three Different Genetic Disorders Causing Intellectual Disability
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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.
KeywordsParental well-being Down syndrome Rett syndrome SF-12 Intellectual disability Genetic disorder
We would like to express our sincere appreciation to all the families who have participated in the Western Australian (WA) Down Syndrome ‘Needs Opinions Wishes’ (NOW) Database, the Australian Rett Syndrome Database (ARSD) and the International CDKL5 Disorder Database (ICDD). We would also like to thank the Australian Surveillance Unit and the Rett Syndrome Association of Australia for their ongoing support in case ascertainment of the ARSD. The 2004 study of the Western Australian (WA) Down Syndrome ‘Needs Opinions Wishes’ (NOW) Database was funded by National Institutes of Health (5R01HD43100-04) and supported by the industry partners; Down syndrome WA, the Disability Services Commission, Edge Employment Solutions and the Department of Education WA. The research projects of the Australian Rett Syndrome Database (ARSD) have previously been funded by the National Institutes of Health (5R01HD043100-05) and the National Health and Medical Research Council project grants (#303189 and #1004384) and program grant (#572742). The management of the International CDKL5 Disorder Database (ICDD) has been supported by the International Foundation for CDKL5 Research. None of the funding bodies has been involved in the study design, data collection and analysis, writing of the article and decision to submit it for publication.
HL and JD provided access to study data; YM, JD, JH and HL conceived and designed the research plan; YM undertook the analysis with input from KW; all authors interpreted the data; YM drafted the paper and all authors edited and read the manuscript and agreed to it being submitted for publication.
Compliance with Ethical Standards
Conflict of interest
The authors declare that they have no conflict of interest.
All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards.
Informed consent was obtained from all individual participants included in the study.
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