Evidence map›Paper›PMID 36974468›Full record

ArticleOphthalmic genetics2023

The validation of inherited retinal disease-specific patient-reported outcome measures in adolescent patients.

Kavin Selvan, Rebhi Abuzaitoun, Maria Fernanda Abalem, Ajoy Vincent, Chris A Andrews, Gabrielle D Lacy, Rafid Farjo, Karissa Kao, Krystal Kao, Gislin Dagnelie and 3 more

Open access · hybridAbstract read
In one paragraph

Article in Ophthalmic genetics, 2023. 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
1.3field-weighted citation impact, top 20% 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

4 citing papers in PubMed, 6 citations in OpenAlex.

  1. Article
  2. Article
  3. Article
  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

13 authors at 4 institutions in 3 countries.

Kavin SelvanGenetics and Genome Biology (GGB) Program, The Hospital for Sick Children Research Institute, Toronto, Ontario, Canada.ORCID 0000-0002-2051-9577
Rebhi AbuzaitounDepartment of Ophthalmology and Visual Sciences, University of Michigan Medical School, Ann Arbor, Michigan, USA.
Maria Fernanda AbalemDepartment of Ophthalmology and Visual Sciences, University of Michigan Medical School, Ann Arbor, Michigan, USA.ORCID 0000-0002-4194-9314
Ajoy VincentGenetics and Genome Biology (GGB) Program, The Hospital for Sick Children Research Institute, Toronto, Ontario, Canada.ORCID 0000-0001-6446-3846
Chris A AndrewsDepartment of Ophthalmology and Visual Sciences, University of Michigan Medical School, Ann Arbor, Michigan, USA.ORCID 0000-0001-5130-6374
Gabrielle D LacyDepartment of Ophthalmology and Visual Sciences, University of Michigan Medical School, Ann Arbor, Michigan, USA.ORCID 0000-0002-3746-9326
Rafid FarjoDepartment of Ophthalmology and Visual Sciences, University of Michigan Medical School, Ann Arbor, Michigan, USA.
Karissa KaoDepartment of Ophthalmology and Visual Sciences, University of Michigan Medical School, Ann Arbor, Michigan, USA.
Krystal KaoDepartment of Ophthalmology and Visual Sciences, University of Michigan Medical School, Ann Arbor, Michigan, USA.
Gislin DagnelieWilmer Eye Institute, Johns Hopkins University School of Medicine, Baltimore, Maryland, USA.ORCID 0000-0001-6707-9735
David C MuschDepartment of Ophthalmology and Visual Sciences, University of Michigan Medical School, Ann Arbor, Michigan, USA.ORCID 0000-0002-4164-3841
K Thiran JayasunderaDepartment of Ophthalmology and Visual Sciences, University of Michigan Medical School, Ann Arbor, Michigan, USA.ORCID 0000-0003-4895-4818
Elise HéonGenetics and Genome Biology (GGB) Program, The Hospital for Sick Children Research Institute, Toronto, Ontario, Canada.ORCID 0000-0003-4143-9311
University of Michigan · USUniversity of Toronto · CAJohns Hopkins University · USUniversidade de São Paulo · BR

Funding

NRSA Training CoreTL1TR002242 · NCATS · UNIVERSITY OF MICHIGAN AT ANN ARBOR · PI HOLINSTAT, MICHAEL ALLAN · 2017 to 2021
$2.8M
Targeted functional testing for retinal diseases based on patient reported outcomesK23EY026985 · NEI · UNIVERSITY OF MICHIGAN AT ANN ARBOR · PI JAYASUNDERA, KANISHKA THIRAN · 2016 to 2020
$1.2M
NCATS NIH HHS TL1 TR002242NEI NIH HHS K23 EY026985
6 · The paper itself

Abstract

purposeTo determine the validity of the validate the adult patient-reported outcome measure tools, the Michigan Retinal Degeneration Questionnaire (MRDQ) and Michigan Vision-Related Anxiety Questionnaire (MVAQ), in adolescent patients with inherited retinal diseases (IRDs).

methodsNinety-one adolescent patients diagnosed with IRDs were recruited at the Hospital for Sick Children (University of Toronto) and the Kellogg Eye Center (University of Michigan). The patients were administered the MRDQ, MVAQ, and Patient Health Questionnaire-4 (PHQ-4). Test-retest variability was assessed in eighteen patients within 14 days of the initial administration. Adolescent responses were analyzed for validity and reliability. As a further validation step, comparisons were made to adult data from the original MRDQ and MVAQ studies to ensure consistency in response ranges.

resultsThe existing MRDQ and MVAQ content and format could accurately detect the impact of IRD on activities of daily living in adolescents with IRDs. No floor/ceiling effects were identified, test-retest reliability was established (r = 0.73-0.86), and no items were excluded after differential item functioning analysis. Domain and trait associations with visual acuity and IRD phenotypes were similar between adolescents and adults.

conclusionsThe MRDQ and MVAQ are psychometrically validated questionnaires for which we have shown validity for use in adolescent patients with IRDs.

Indexed as

Activities of Daily LivingRetinal DegenerationHumansPatient Reported Outcome MeasuresPsychometricsReproducibility of ResultsSurveys and Questionnairesclinical trialgene therapyinherited retinal diseasePatient-reported outcomespediatricsPROMretinal dystrophy

Identifiers

PMID36974468
PMCPMC10198816
OpenAlexW4321523610

What OpenQuestion holds

Textmetadata
LicenceTDM
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.