Evidence map›Paper›PMID 38811427›Full record

ArticleJournal of patient-reported outcomes2024

Quality of life in individuals with neurofibromatosis type 1 associated cutaneous neurofibromas: validation of the Dutch cNF-Skindex.

Britt A E Dhaenens, Sarah A van Dijk, Laura Fertitta, Walter Taal, Pierre Wolkenstein, Rianne Oostenbrink

Abstract readValidation Study
In one paragraph

Article in Journal of patient-reported outcomes, 2024. 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
–field-weighted citation impact
1 · What the graph read from 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.

2 · The registry

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3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

  1. 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

6 authors.

Britt A E DhaenensDepartment of General Paediatrics, Erasmus MC-Sophia Children's Hospital, Wytemaweg 80, Rotterdam, 3015 CN, The Netherlands. b.dhaenens@erasmusmc.nl.ORCID 0000-0003-0928-4049
Sarah A van DijkThe ENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus MC, Rotterdam, The Netherlands.ORCID 0009-0002-2851-4620
Laura FertittaDepartment of Dermatology, National Referral Center for Neurofibromatosis, Henri Mondor University Hospital, Assistance Publique- Hôpitaux de Paris, Creteil, France.ORCID 0000-0003-3528-9804
Walter TaalThe ENCORE Expertise Centre for Neurodevelopmental Disorders, Erasmus MC, Rotterdam, The Netherlands.ORCID 0000-0002-5530-7259
Pierre WolkensteinDepartment of Dermatology, National Referral Center for Neurofibromatosis, Henri Mondor University Hospital, Assistance Publique- Hôpitaux de Paris, Creteil, France.ORCID 0000-0001-8194-457X
Rianne OostenbrinkDepartment of General Paediatrics, Erasmus MC-Sophia Children's Hospital, Wytemaweg 80, Rotterdam, 3015 CN, The Netherlands.ORCID 0000-0001-7919-8934

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

backgroundAlmost all patients with Neurofibromatosis type 1 (NF1) develop cutaneous neurofibroma (cNF), benign dermal tumours that have a large impact on the patient's Quality of Life (QoL). The French cNF-Skindex is the first questionnaire to specifically assess cNF-related QoL in patients with NF1. We aimed to adapt and validate a Dutch version of the cNF-Skindex.

methodsThe questionnaire was translated using forward and backwards translation, and subsequently administered to a sample of 59 patients on two separate occasions. Feasibility was evaluated by the presence of floor/ceiling effects. Reliability was assessed by evaluating internal consistency and test-retest reliability, by calculating Cronbach's alpha and Spearman's rank correlation coefficients. The EQ-5D-5L and SF-36 were used to evaluate convergent validity, using Spearman's rank correlation coefficients. An exploratory factor analysis was performed to study the data's internal structure. Multivariable linear regression was used to model the relationship between patient characteristics and the cNF-Skindex.

resultsThe Dutch cNF-Skindex demonstrated excellent feasibility and reliability (Cronbach's alpha 0.96, test-retest correlation coefficient 0.88). Convergent validity was confirmed for the EQ-5D-5L and relevant SF-36 scales. All items and subdomains from the original questionnaire were confirmed following exploratory factor analysis. The patient characteristics included in the multivariable linear regression were not significantly associated with the cNF-Skindex score.

conclusionsThe Dutch cNF-Skindex displayed excellent psychometric properties, enabling use in the Netherlands.

Indexed as

NeurofibromaNeurofibromatosis 1Quality of LifeSkin NeoplasmsAdolescentAdultFemaleHumansMaleMiddle AgedNetherlandsPsychometricsReproducibility of ResultsSurveys and QuestionnairesYoung AdultcNF-SkindexCutaneous neurofibromaNeurofibromatosis type 1Patient-reported outcome measureQoLQuality of life

Identifiers

PMID38811427
PMCPMC11136889

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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.