Evidence map›Paper›PMID 31257692›Full record

ReviewJournal of paediatrics and child health2019

Requirements for improving health and well-being of children with Prader-Willi syndrome and their families.

Jessica Mackay, Zoe McCallum, Geoffrey R Ambler, Komal Vora, Gillian Nixon, Philip Bergman, Nora Shields, Kate Milner, Nitin Kapur, Patricia Crock and 16 more

Open access · bronzeAbstract readReview
PubMed Full text at PMC Publisher
In one paragraph

Review in Journal of paediatrics and child health, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 9 papers, 1 of them a synthesis that pooled it.

0numbers the graph read from it
0cells of the map it votes in
9citing papers in PubMed, 1 pooled it
1.4field-weighted citation impact, top 17% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

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.

2 · The registry

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.

3 · Its place in the literature

Who cites it

9 citing papers in PubMed, 1 synthesis or guideline pooled it, 16 citations in OpenAlex.

  1. Pooled it
  2. Article
  3. Review
  4. Article
  5. Article
  6. Article
  7. Article
  8. Article
  9. Review
4 · The record

Corrections and comments

PubMed lists nothing against this paper. Absence here is not a guarantee, only a check that was made.

5 · Who and what money

Authors and funding

26 authors at 17 institutions in 3 countries.

Jessica MackayTelethon Kids Institute, Centre for Child Health Research, University of Western Australia, Perth, Western Australia, Australia.
Zoe McCallumDepartment of Neurodevelopment and Disability, Royal Children's Hospital, Melbourne, Victoria, Australia.
Geoffrey R AmblerInstitute of Endocrinology and Diabetes, Children's Hospital at Westmead, Sydney, New South Wales, Australia.
Komal VoraDepartment of Paediatric Endocrinology and Diabetes, John Hunter Children's Hospital, Newcastle, New South Wales, Australia.
Gillian NixonMelbourne Children's Sleep Centre, Monash Children's Hospital, Melbourne, Victoria, Australia.
Philip BergmanDepartment of Paediatrics, Monash University, Melbourne, Victoria, Australia.
Nora ShieldsSchool of Allied Health, La Trobe University, Melbourne, Victoria, Australia.
Kate MilnerDepartment of Neurodevelopment and Disability, Royal Children's Hospital, Melbourne, Victoria, Australia.
Nitin KapurRespiratory and Sleep Medicine, Queensland Children's Hospital, Brisbane, Queensland, Australia.
Patricia CrockDepartment of Paediatric Endocrinology and Diabetes, John Hunter Children's Hospital, Newcastle, New South Wales, Australia.
Daan CaudriTelethon Kids Institute, Centre for Child Health Research, University of Western Australia, Perth, Western Australia, Australia.
Jaqueline CurranDepartment of Endocrinology, Perth Children's Hospital, Perth, Western Australia, Australia.
Charles VergeDepartment of Endocrinology, Sydney Children's Hospital, Sydney, New South Wales, Australia.
Chris SetonDepartment of Sleep Medicine, Children's Hospital Westmead, Sydney, New South Wales, Australia.
Andrew TaiRespiratory and Sleep Department, Women's and Children's Hospital, Adelaide, South Australia, Australia.
Elaine ThamEndrocrinology and Diabetes Department, Women's and Children's Hospital, Adelaide, South Australia, Australia.
Yassmin MusthaffaDiamantina Institute, Faculty of Medicine, University of Queensland, Brisbane, Queensland, Australia.
Antony R LaffertyDepartment of Endocrinology and Diabetes, Canberra Hospital, Canberra, Australian Capital Territory, Australia.
Greg BlecherDepartment of Sleep Medicine, Sydney Children's Hospital, Sydney, New South Wales, Australia.
Jessica HarperDepartment of Endocrinology, Sydney Children's Hospital, Sydney, New South Wales, Australia.
Cara SchofieldTelethon Kids Institute, Centre for Child Health Research, University of Western Australia, Perth, Western Australia, Australia.
Aleisha NielsenRespiratory and Sleep Medicine, Perth Children's Hospital, Perth, Western Australia, Australia.
Andrew WilsonTelethon Kids Institute, Centre for Child Health Research, University of Western Australia, Perth, Western Australia, Australia.
Helen LeonardTelethon Kids Institute, Centre for Child Health Research, University of Western Australia, Perth, Western Australia, Australia.
Catherine S ChoongTelethon Kids Institute, Centre for Child Health Research, University of Western Australia, Perth, Western Australia, Australia.
Jenny DownsTelethon Kids Institute, Centre for Child Health Research, University of Western Australia, Perth, Western Australia, Australia.ORCID https://orcid.org/0000-0001-7358-9037
The Kids Research Institute Australia · AUPrincess Margaret Hospital for Children · AURoyal Children's Hospital · AUWomen's and Children's Hospital · AUAustralian National University · AUBoston Children's Hospital · USChildren's Hospital at Westmead · AUHudson Institute of Medical Research · AUHunter Medical Research Institute · AUJohn Hunter Children's Hospital · AULa Trobe University · AUMonash Children’s Hospital · AUSydney Children's Hospital · AUThe University of Queensland · AUThe University of Sydney · AUTranslational Research Institute · AUUNSW Sydney · AU

Funding

Perth Children's Hospital FoundationPrader Willi Association USA
6 · The paper itself

Abstract

Prader-Willi syndrome (PWS) is a rare genetic condition with multi-system involvement. The literature was reviewed to describe neurodevelopment and the behavioural phenotype, endocrine and metabolic disorders and respiratory and sleep functioning. Implications for child and family quality of life were explored. Challenging behaviours contribute to poorer well-being and quality of life for both the child and caregiver. Recent evidence indicates healthy outcomes of weight and height can be achieved with growth hormone therapy and dietary restriction and should be the current target for all individuals with PWS. Gaps in the literature included therapies to manage challenging behaviours, as well as understanding the effects of growth hormone on respiratory and sleep function. New knowledge regarding the transition of children and families from schooling and paediatric health services to employment, accommodation and adult health services is also needed. Developing a national population-based registry could address these knowledge gaps and inform advocacy for support services that improve the well-being of individuals with PWS and their families.

Indexed as

Personal SatisfactionQuality of LifeAdolescentChildChild, PreschoolFamilyHumansHyperphagiaPrader-Willi SyndromeendocrinehyperphagiaPrader-Willi syndromequality of lifesleep disordered breathing

Identifiers

PMID31257692
PMCPMC6852695
OpenAlexW2953482367

What OpenQuestion holds

Textmetadata
LicenceCC BY-NC-ND
Read underepoch 390

Registered trials

None linked

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.