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Title: Parental health spillover effects of paediatric rare genetic conditions.
Austin Authors: Wu, You;Al-Janabi, Hareth;Mallett, Andrew;Quinlan, Catherine;Scheffer, Ingrid E ;Howell, Katherine B;Christodoulou, John;Leventer, Richard J;Lockhart, Paul J;Stark, Zornitza;Boughtwood, Tiffany;Goranitis, Ilias
Affiliation: Disciplines of Genetic Medicine and Child and Adolescent Health, Sydney Medical School, University of Sydney, Sydney, NSW, Australia
Department of Medicine, Austin Health, The University of Melbourne, Heidelberg, Victoria, Australia
Department of Neurology, Royal Children's Hospital, Melbourne, VIC, Australia
Institute of Applied Health Research, University of Birmingham, Birmingham, UK
Centre for Health Policy, University of Melbourne, Melbourne, VIC, Australia
Australian Genomics Health Alliance, Melbourne, VIC, Australia
Murdoch Children's Research Institute, Melbourne, VIC, Australia
Kidney Health Service and Conjoint Renal Research Laboratory, Royal Brisbane and Women's Hospital, Brisbane, QLD, Australia
Institute for Molecular Bioscience and Faculty of Medicine, The University of Queensland, Brisbane, QLD, Australia
Department of Paediatric Nephrology, Royal Children's Hospital, Melbourne, VIC, Australia
Department of Paediatrics, University of Melbourne, Melbourne, VIC, Australia
Issue Date: Sep-2020 2020-04-07
Publication information: Quality of Life Research 2020; 29(9): 2445-2454
Abstract: The complexity and severity of rare genetic conditions pose substantial burden to families. While the importance of spillovers on carers' health in resource allocation decisions is increasingly recognised, there is significant lack of empirical evidence in the context of rare diseases. The objective of this study was to estimate the health spillovers of paediatric rare genetic conditions on parents. Health-related quality-of-life (HRQoL) data from children with rare genetic conditions (genetic kidney diseases, mitochondrial diseases, epileptic encephalopathies, brain malformations) and their parents were collected using the CHU9D and SF-12 measures, respectively. We used two approaches to estimate parental health spillovers. To quantify the 'absolute health spillover', we matched our parent cohort to the Australian general population. To quantify the 'relative health spillover', regression models were applied using the cohort data. Parents of affected children had significantly lower HRQoL compared to matched parents in the general public (- 0.06; 95% CIs - 0.08, - 0.04). Multivariable regression demonstrated a positive association between parental and child health. The mean magnitude of HRQoL loss in parents was estimated to be 33% of the HRQoL loss observed in children (95% CIs 21%, 46%). Paediatric rare genetic conditions appear to be associated with substantial parental health spillovers. This highlights the importance of including health effects on family members and caregivers into economic evaluation of genomic technologies and personalised medicine. Overlooking spillover effects may undervalue the benefits of diagnosis and management in this context. This study also expands the knowledge of family spillover to the rare disease spectrum.
DOI: 10.1007/s11136-020-02497-3
ORCID: 0000-0002-2311-2174
Journal: Quality of Life Research
PubMed URL: 32266555
Type: Journal Article
Subjects: Economic evaluation
Genomic medicine
Informal care
Quality of life
Rare disease
Appears in Collections:Journal articles

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