Evidence map›Paper›PMID 40471421›Full record

ArticleJournal of neuro-oncology2025

Psychosocial functioning and determinants of the health-related quality of life in children with neurofibromatosis type 1 and cognitive impairments.

Belinda Barton, Pamela L Wolters, Karin S Walsh, Nicole J Ullrich, Tena Rosser, James Tonsgard, David Viskochil, Elizabeth Schorry, Laura J Klesse, Michael J Fisher and 7 more

Abstract read
In one paragraph

Article in Journal of neuro-oncology, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 4 papers.

0numbers the graph read from it
0cells of the map it votes in
4citing papers in PubMed
–field-weighted citation impact
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

4 citing papers in PubMed.

  1. Article
  2. Article
  3. Review
  4. 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

17 authors.

Belinda BartonFaculty of Health, Southern Cross University, Coffs Harbour, NSW, Australia. belinda.barton@scu.edu.au.ORCID https://orcid.org/0000-0002-3157-2031
Pamela L WoltersPediatric Oncology Branch, Center for Cancer Research, National Cancer Institute, National Institutes of Health, Bethesda, MD, USA.ORCID https://orcid.org/0000-0001-9319-6281
Karin S WalshCenter for Neuroscience and Behavioral Medicine, Division of Neuropsychology, Children's National Hospital, Washington, DC, USA.ORCID https://orcid.org/0000-0001-9637-2164
Nicole J UllrichDepartment of Neurology, Boston Children's Hospital, Boston, MA, USA.
Tena RosserDepartment of Neurology, Children's Hospital of Los Angeles, Los Angeles, CA, USA.
James TonsgardDivision of Neurology, University of Chicago Medicine Comer Children's Hospital, Chicago, IL, USA.
David ViskochilDepartment of Genetics, University of Utah, Salt Lake City, USA.
Elizabeth SchorryHuman Genetics, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, USA.
Laura J KlesseDepartment of Pediatrics, University of Texas Southwestern Medical Center, Dallas, USA.ORCID https://orcid.org/0000-0003-1323-7720
Michael J FisherDivision of Oncology, The Children's Hospital of Philadelphia, Philadelphia, PA, USA.
David H GutmannDepartment of Neurology, Washington University School of Medicine, St Louis, MO, USA.
Roger J PackerGilbert Family Neurofibromatosis Institute and Brain Tumor Institute, Children's National Hospital, Washington, DC, USA.
Bruce KorfDepartment of Genetics, University of Alabama at Birmingham, Birmingham, USA.
Maria T AcostaUndiagnosed Disease Program, Office of the Clinical Director, National Human Genome Research Institute, National Institute of Health, Bethesda, MD, USA.ORCID https://orcid.org/0000-0001-8914-7401
Kathryn N NorthMurdoch Children's Research Institute, The Royal Children's Hospital, Parkville, VIC, Australia.
Jonathan M PayneMurdoch Children's Research Institute, The Royal Children's Hospital, Parkville, VIC, Australia.ORCID https://orcid.org/0000-0001-9565-3845
NF Clinical Trials Consortium

Funding

CTSA UM1 Program at University of UtahUM1TR004409 · NCATS · UTAH STATE HIGHER EDUCATION SYSTEM--UNIVERSITY OF UTAH · PI RACHEL HESS, Jennifer Juhl Majersik · 2023 to 2026
$21.9M
Genetic Analysis and Manipulation Core (GAEC)P50HD105351 · NICHD · BOSTON CHILDREN'S HOSPITAL · PI Hisashi Umemori · 2021 to 2026
$9.4M
NCATS NIH HHS UM1 TR004409NICHD NIH HHS P50 HD105351
6 · The paper itself

Abstract

purposeNeurofibromatosis type 1 (NF1) is an autosomal dominant genetic condition associated with cutaneous and neoplastic manifestations and other physical manifestations, as well as cognitive, psychosocial, and behavioural difficulties. NF1 negatively impacts the health-related quality of life (HRQoL) of children. There is limited evidence regarding the determinants of HRQoL of children with NF1. The aim of this study was to (i) compare the HRQoL of children with NF1 and cognitive impairments to published data of healthy children and children with cancer and (ii) identify specific determinants of child and parent-proxy reports of psychosocial HRQoL.

methodsChildren with NF1 and cognitive impairments (n = 135, 8-15 years 11 months) and their parents completed standardized measures assessing children's HRQoL, behavioral and emotional functioning. Children completed a brief intelligence test. Correlations and multiple linear regressions were conducted to identify determinants of psychosocial HRQoL.

resultsChildren with NF1 had significantly poorer HRQoL for all domains than published data of healthy children and significantly poorer HRQoL for Psychosocial Health, School and Social Functioning than published data of children with cancer. For child self-report, attention problems and increased social stress predicted their psychosocial HRQoL. For parent-proxy reports, activities of daily living and depression were significant predictors of children's psychosocial HRQoL. Social stress and depression were the strongest predictors of Psychosocial HRQoL.

conclusionRoutine screening and early identification of depressive symptoms and interventions that promote social support, coping and resiliency may improve the HRQoL of children with NF1.

Indexed as

Cognitive DysfunctionNeurofibromatosis 1Psychosocial FunctioningQuality of LifeActivities of Daily LivingAdolescentChildFemaleHumansMaleParentsChildrenDepressionNeurofibromatosis type 1PsychosocialQuality of lifeSocial stress

Identifiers

PMID40471421
PMCPMC12198070

What OpenQuestion holds

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LicenceCC BY
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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.