ArticleQuality of life research : an international journal of quality of life aspects of treatment, care and rehabilitation2020
Parental health spillover effects of paediatric rare genetic conditions.
Article in Quality of life research : an international journal of quality of life aspects of treatment, care and rehabilitation, 2020. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 33 papers, 7 of them syntheses that pooled it.
What it found
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The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.
The trial behind it
Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.
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Who cites it
33 citing papers in PubMed, 7 syntheses or guidelines pooled it.
- Clinical and cost-effectiveness of detailed anomaly ultrasound screening in the first trimester: a mixed-methods study.Health technology assessment (Winchester, England) · 2025Pooled it
- A Systematic Review of Methods Used by Pediatric Cost-Utility Analyses to Include Family Spillover Effects.PharmacoEconomics · 2024Pooled it
- The psychosocial impact of childhood dementia on children and their parents: a systematic review.Orphanet journal of rare diseases · 2023Pooled it
- The psychosocial impact of childhood dementia on children and their parents: a systematic review.Orphanet journal of rare diseases · 2023Pooled it
- A systematic review of economic evaluations forInternational journal of technology assessment in health care · 2023Pooled it
- Perception of Social and Educational Quality of Life of Minors Diagnosed with Rare Diseases: A Systematic Review and Meta-Analysis.International journal of environmental research and public health · 2023Pooled it
- Principles of Genomic Newborn Screening Programs: A Systematic Review.JAMA network open · 2021Pooled it
- Recommendations for the use of clinical outcome assessments in rare disease drug development.EClinicalMedicine · 2026Review
- Content validity, face validity and comprehensiveness of generic quality-of-life measures in adults and children with rare genetic conditions and their carers: a think aloud qualitative study.Quality of life research : an international journal of quality of life aspects of treatment, care and rehabilitation · 2026Article
- Exploring relative health spillover effects within the context of dementia using the EQ-HWB-9.Health and quality of life outcomes · 2026Article
- Measuring health spillover effects in household members of adolescents and elderly individuals with health issues in China.Quality of life research : an international journal of quality of life aspects of treatment, care and rehabilitation · 2026Article
- Measuring and valuing spillover effects in caregivers and families: A scoping review.PloS one · 2026Article
- Determining the value of genomics in healthcare.Nature medicine · 2025Review
- The psychometric performance of the EQ-HWB-9 for measuring health and wellbeing in a general population sample from Australia and New Zealand.Quality of life research : an international journal of quality of life aspects of treatment, care and rehabilitation · 2025Article
- Stakeholder survey about broad elements of value in health technology assessment in Australia: industry and academia more similar than different.International journal of technology assessment in health care · 2025Article
- A roadmap for genome projects to foster psychosocial and economic evidence to further policy and practice.Communications medicine · 2025Review
- The performance of the EQ-HWB-S as a measure of quality-of-life of caregivers in families that have experienced adverse events.The European journal of health economics : HEPAC : health economics in prevention and care · 2025Article
- On spillovers in economic evaluations: definition, mapping review and research agenda.The European journal of health economics : HEPAC : health economics in prevention and care · 2024Review
- Experiences of quality of life and access to health services among rare disease caregivers: a scoping review.Orphanet journal of rare diseases · 2024Article
- Bayesian cost-effectiveness analysis of Whole genome sequencing versus Whole exome sequencing in a pediatric population with suspected genetic disorders.The European journal of health economics : HEPAC : health economics in prevention and care · 2024Article
Corrections and comments
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Authors and funding
12 authors.
Funding
Abstract
purposeThe 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.
methodsHealth-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.
resultsParents 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%).
conclusionPaediatric 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.
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Registered trials
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