Evidence map›Paper›PMID 33748828›Full record

ArticleJournal of human & clinical genetics2019

HRQoL in Barth Syndrome: Agreement between Child Self-reports and Parent Proxy-reports and Its Relationship to Parental HRQoL.

Yoonjeong Lim, Consuelo M Kreider, Mary Alvarez, Roxanna M Bendixen

Open access · diamondAbstract read
PubMed Full text at PMC Publisher
In one paragraph

Article in Journal of human & clinical genetics, 2019. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
0.7field-weighted citation impact, top 23% 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

1 citing paper in PubMed, 6 citations in OpenAlex.

  1. Barth Syndrome: Psychosocial Impact and Quality of Life Assessment.Journal of cardiovascular development and disease · 2022
    Article
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

4 authors at 3 institutions in 1 country.

Yoonjeong LimDepartment of Occupational Therapy Byrdine F. Lewis College of Nursing & Health Professions, Georgia State University, Atlanta, GA, USA.
Consuelo M KreiderDepartment of Occupational Therapy, College of Public Health and Health Professions, University of Florida, Gainesville, FL, USA.
Mary AlvarezDepartment of Occupational Therapy Byrdine F. Lewis College of Nursing & Health Professions, Georgia State University, Atlanta, GA, USA.
Roxanna M BendixenDepartment of Occupational Therapy, School of Health and Rehabilitation Sciences, University of Pittsburgh, Pittsburgh, PA, USA.
Georgia State University · USUniversity of Florida · USUniversity of Pittsburgh · US

Funding

Rehabilitation Research Career Development ProgramK12HD055929 · NICHD · UNIVERSITY OF TEXAS MED BR GALVESTON · PI BROWN, DAVID A · 2007 to 2021
$11.7M
Comprehensive Assessment of the Impact of Illness and Disability in ChildrenK01HD064778 · NICHD · UNIVERSITY OF PITTSBURGH AT PITTSBURGH · PI BENDIXEN, ROXANNA MARIE · 2010 to 2014
$551k
NICHD NIH HHS K01 HD064778NICHD NIH HHS K12 HD055929
6 · The paper itself

Abstract

purposeBarth syndrome is an X-linked rare disorder that typically affects only males. This study investigates 1) agreement between child self-reports and parent proxy-reports of HRQoL in boys with Barth syndrome and 2) relationship between parental HRQoL and parent proxy-reports of HRQoL for the child. MATERIALS AND

methodsTwenty-eight boys with Barth syndrome and their parents participated in this study. The PedsQL™ 4.0 and the PedsQL™ Family Impact Module were used to measure HRQoL of the boys, and the parents' HRQoL, respectively. The Intraclass Correlation Coefficient was used to test agreement between the child self-reports and parent proxy-reports of HRQoL. The Spearman correlation coefficient was used to test the relationship between parental HRQoL and parent proxy-reports of HRQoL for the child.

resultsThe agreement between the child self-reports and the parent proxy-reports showed moderate-to-good agreement. Higher parental HRQoL was significantly related to higher ratings of the parents on their children's HRQoL (

conclusionsThis study broadens understanding of HRQoL of boys with Barth syndrome using both child self-reports and parent proxy-reports. The findings indicate that the parent proxy-report of HRQoL should be used in conjunction with the child self-report when making client-centered health decisions.

Indexed as

Barth síndromeHealth-related quality of lifeHRQoLProxy-reportRare DiseasesSelf-report

Identifiers

PMID33748828
PMCPMC7971423
OpenAlexW3000192889

What OpenQuestion holds

Textmetadata
LicenceCC BY
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.