Parental health spillover effects of paediatric rare genetic conditions
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Parental health spillover effects of paediatric rare genetic conditions You Wu1,2,3 · Hareth Al‑Janabi4 · Andrew Mallett2,3,5,6 · Catherine Quinlan2,3,7,8 · Ingrid E. Scheffer2,3,8,9,10 · Katherine B. Howell3,8,10 · John Christodoulou2,3,8,11 · Richard J. Leventer2,3,8,10 · Paul J. Lockhart2,3,8 · Zornitza Stark2,3,8 · Tiffany Boughtwood2,3 · Ilias Goranitis1,2,3 Accepted: 31 March 2020 © Springer Nature Switzerland AG 2020
Abstract Purpose 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. Methods 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. Results 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%). Conclusion 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. Keywords Rare disease · Quality of life · Economic evaluation · Informal care · Genomic medicine
Electronic supplementary material The online version of this article (https://doi.org/10.1007/s11136-020-02497-3) contains supplementary material, which is available to authorized users. * Ilias Goranitis [email protected] 1
Centre for Health Policy, University of Melbourne, Melbourne, VIC, Australia
2
Australian Genomics Health Alliance, Melbourne, VIC, Australia
3
Murdoch Children’s Research Institute, Melbourne, VIC, Australia
4
Institute of Applied Health Research, University of Birmingham, Birmingham, UK
5
Kidney Health Service and Conjoint Renal Research Laboratory, Royal Brisbane and Women’s Hospital, Brisbane, QLD, Australia
6
Institute for Molecular Bioscience a
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