ArticlePloS one2018
High levels of caregiver burden in Prader-Willi syndrome.
Article in PloS one, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 58 papers, 3 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.
Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.
Who cites it
58 citing papers in PubMed, 3 syntheses or guidelines pooled it, 104 citations in OpenAlex.
- The burden of illness in Prader-Willi syndrome: a systematic literature review.Orphanet journal of rare diseases · 2025Pooled it
- Effects of probiotics on patients with Prader-Willi syndrome: a systematic review and meta-analysis of randomized controlled trials.Frontiers in nutrition · 2025Pooled it
- Resilience in Familial Caregivers of Children with Developmental Disabilities: A Meta-analysis.Journal of autism and developmental disorders · 2020Pooled it
- Intranasal Carbetocin Reduces Hyperphagia, Anxiousness, and Distress in Prader-Willi Syndrome: CARE-PWS Phase 3 Trial.The Journal of clinical endocrinology and metabolism · 2023Trial
- Quality of life outcomes in two phase 3 trials of setmelanotide in patients with obesity due to LEPR or POMC deficiency.Orphanet journal of rare diseases · 2022Trial
- Obstructive sleep apnoea severity and insulin resistance in children with Prader-Willi Syndrome: the SPISE index as a candidate metabolic marker.European journal of pediatrics · 2026Article
- Hyperphagia in Prader-Willi syndrome: linking hypothalamic dysfunction to clinical assessment and management across the lifespan.Orphanet journal of rare diseases · 2026Review
- Behavior Regulation Abilities in Children With Prader-Willi Syndrome and Their Impact on Daily Life: At the Crossroads of Cognitive and Emotional Development.Journal of autism and developmental disorders · 2026Article
- Low dose systemic AAV-exendin-4 gene therapy for Prader-Willi syndrome and dietary obesity.Molecular therapy. Advances · 2026Article
- Lived Experiences of Teens and Adults in North America Who Grew Up With a Sibling Diagnosed With Prader Willi Syndrome.Journal of applied research in intellectual disabilities : JARID · 2026Article
- Social support and maternal caregiving burden in families of children with Angelman syndrome in China: the mediating role of self-stigma.BMC psychology · 2026Article
- Patient advocacy group perspectives on treatment priorities and clinical trials for the rare neurodevelopmental condition, Prader-Willi syndrome.Orphanet journal of rare diseases · 2026Article
- Efficacy and safety of semaglutide for obesity and hyperphagia in adults with Prader-Willi syndrome.Frontiers in endocrinology · 2026Observational
- Patient-centered burdens and economic outcomes among patients who are veterans, people with intellectual and developmental disabilities, and people living in rural areas and their caregivers: a scoping review.Frontiers in public health · 2026Article
- Codesigning a Neurocognitive Assessment Protocol for Hyperphagia: Perspectives From Stakeholders in Prader-Willi Syndrome.Journal of intellectual disability research : JIDR · 2026Article
- Fiber Intervention Study in Prader-Willi Syndrome: Insights into Metabolic and Microbiota Shifts.The Journal of clinical endocrinology and metabolism · 2025Article
- The relationship between stress, anxiety, and health literacy in parents of children with chronic gastroenterological diseases: a multi-center cross-sectional study.BMC public health · 2025Article
- Anxiety, Depression and Stress in Parents and Siblings of People Who Have Prader-Willi Syndrome: Morbidity Prevalence and Mitigating Factors.Journal of intellectual disability research : JIDR · 2025Article
- Validation of the Food Safe Zone questionnaire for families of individuals with Prader-Willi syndrome.Journal of neurodevelopmental disorders · 2025Article
- Environmental Enrichment Normalizes Metabolic Function in the Murine Model of Prader-Willi Syndrome Magel2-Null Mice.Endocrinology · 2025Article
Corrections and comments
PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.
Authors and funding
5 authors at 3 institutions in 1 country.
Funding
No grant is acknowledged in the PubMed record.
Abstract
objectivesPrader-Willi syndrome (PWS) is a rare genetic neurodevelopmental disorder that is characterized by hyperphagia, developmental delay, incomplete sexual development, mild-to-moderate intellectual disability, and a variety of challenging behavioral and psychiatric symptoms. The characteristics of PWS can be difficult for caregivers to cope with and are likely to cause significant and long- term caregiver burden. The current study examined burden in 142 caregivers of children and adults with PWS living in the US using the Zarit Burden Interview (ZBI). The study aimed to measure the level of burden in caregivers of individuals with PWS, to explore the impact of PWS on caregiver quality of life, and to assess ZBI as an indicator of that impact.
resultsCaregivers participating in this study were predominantly mothers, 30-59 years old, non-Hispanic Whites, married or in a relationship, with an annual household income slightly distributed towards higher income. Nearly 90% of the caregiver`s children with PWS lived at home. Caregivers experienced high caregiver burden with an average ZBI score of 44.4 ± 15.4. ZBI scores were highest for caregivers of teenage and young adult individuals with PWS (49.2 ± 14.6 and 49.2 ± 14.1, respectively), while those caring for older adults (>30) and the youngest age group had lower scores (38.6 ±10.5 and 34.8 ±12.5, respectively). Caregivers reported that caring for a person with PWS negatively impacted their romantic relationship, ability to work, sleep, and mood. Whereas we did not find strong correlations between family income or level of help the caregiver receives and ZBI scores, the results showed significant correlations and a linear relationship between ZBI scores and caregiver depressed mood, feelings of anxiety, negative romantic relationship impact, as well as sleep and work disruption.
conclusionsOur study reveals that PWS incurs high caregiver burden and impacts many aspects of the lives of caregiver. We identified the ZBI as a good predictor of that impact. Our findings draw attention to the critical unmet need for support for caregivers of individuals with PWS.
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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.