Evidence map›Paper›PMID 37183616›Full record

ArticleJournal of genetic counseling2023

Parent perspectives on disclosing a pediatric neurofibromatosis type 1 diagnosis.

Madeleine Franchi, Heather B Radtke, Andrea M Lewis, Irene Moss, Stacey S Cofield, Ashley Cannon

Open access · hybridAbstract read
In one paragraph

Article in Journal of genetic counseling, 2023. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Not yet cited in PubMed.

0numbers the graph read from it
0cells of the map it votes in
0citing papers in PubMed
0.2field-weighted citation impact, top 45% 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

0 citing papers in PubMed, 1 citations in OpenAlex.

No citing paper in PubMed yet.

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 at 4 institutions in 1 country.

Madeleine FranchiDepartment of Clinical and Diagnostic Sciences, University of Alabama at Birmingham, Birmingham, Alabama, USA.ORCID 0000-0003-4632-3529
Heather B RadtkeDepartment of Pediatrics, Medical College of Wisconsin, Milwaukee, Wisconsin, USA.ORCID 0000-0003-1557-7390
Andrea M LewisInvitae Corporation, San Francisco, California, USA.ORCID 0000-0002-1116-4276
Irene MossDepartment of Genetics, University of Alabama at Birmingham, Birmingham, Alabama, USA.
Stacey S CofieldDepartment of Biostatistics, University of Alabama at Birmingham, Birmingham, Alabama, USA.ORCID 0000-0002-1104-3087
Ashley CannonDepartment of Clinical and Diagnostic Sciences, University of Alabama at Birmingham, Birmingham, Alabama, USA.ORCID 0000-0002-2319-208X
University of Alabama at Birmingham · USInvitae (United States) · USMedical College of Wisconsin · USSurdna Foundation · US

Funding

National Dissemination of I-Corps@NCATS: Accelerating Translation through CommercializationUL1TR003096 · NCATS · UNIVERSITY OF ALABAMA AT BIRMINGHAM · PI GUTIERREZ, ORLANDO M, KIMBERLY, ROBERT P. · 2019 to 2023
$43.6M
NCATS NIH HHS UL1 TR003096
6 · The paper itself

Abstract

Neurofibromatosis 1 (NF1) is a common genetic disorder typically diagnosed in childhood and characterized by cutaneous findings, nerve sheath tumors, skeletal abnormalities, malignancies, and developmental differences. Due to its variability, NF1 is an unpredictable condition that parents have concerns about discussing with their children. While there are publications addressing the disclosure of genetic conditions in general, no NF1-specific disclosure literature exists. To fill this gap, this mixed methods study sought to evaluate the concerns, barriers, failures, or successes parents or guardians have experienced when they have or have not chosen to tell their child(ren) about an NF1 diagnosis. Parents of children between ages 0 and 17 with a diagnosis of NF1 completed a survey and some parents were selected for an interview invitation. A total of 258 surveys were completed, and 20 parents were interviewed. Interview transcripts were categorized into disclosure and non-disclosure groups. Themes were organized into five categories based on interview questions: disclosure concerns, factors affecting disclosure/non-disclosure, approaches to disclosure, desired resources, and recommendations for disclosure. Sentiment analysis was performed on responses about the disclosure discussion itself. Results indicated that most parents (70.5%) disclosed the NF1 diagnosis to their child and overall felt it was a positive experience. Almost one-third of parents (29.5%) had not disclosed the diagnosis. A strong significance was identified between disclosure and severe presentation of NF1 (p = 0.0008). Parents in both groups shared similar concerns about discussing the diagnosis and multiple factors influenced the disclosure decision. Most parents approached disclosure as a process and emphasized the need to be honest and supportive of their child. Parents highlighted the need for more educational resources for children and guidance on how to disclose. These findings indicate that additional resources and support for parents would facilitate disclosure and the involvement of genetic counselors in the process would be beneficial.

Indexed as

Neurofibromatosis 1ParentsAdolescentAdultChildChild, PreschoolFemaleHumansInfantInfant, NewbornMaleSurveys and QuestionnairesTruth Disclosureattitudesgenetic counselingneurofibromatosis type 1parentspediatricspsychosocial

Identifiers

PMID37183616
PMCPMC10592428
OpenAlexW4376614552

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.