Evidence map›Paper›PMID 29914349›Full record

ArticleJournal of neurodevelopmental disorders2018

Social skills in children with RASopathies: a comparison of Noonan syndrome and neurofibromatosis type 1.

Elizabeth I Pierpont, Rebekah L Hudock, Allison M Foy, Margaret Semrud-Clikeman, Mary Ella Pierpont, Susan A Berry, Ryan Shanley, Nathan Rubin, Katherine Sommer, Christopher L Moertel

Open access · goldAbstract readComparative Study
In one paragraph

Article in Journal of neurodevelopmental disorders, 2018. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 26 papers, 5 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
26citing papers in PubMed, 5 pooled it
1.7field-weighted citation impact, top 16% 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

26 citing papers in PubMed, 5 syntheses or guidelines pooled it, 45 citations in OpenAlex.

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  14. Biomarker Landscape in RASopathies.International journal of molecular sciences · 2024
    Review
  15. Article
  16. Neuropsychiatric phenotypes in children with Noonan syndrome.Developmental medicine and child neurology · 2023
    Article
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  18. Article
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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

10 authors at 1 institution in 1 country.

Elizabeth I PierpontDivision of Clinical Behavioral Neuroscience, Department of Pediatrics, University of Minnesota, 420 Delaware Street SE, Mayo Mail Code 486, Minneapolis, MN, 55455, USA. pier0053@umn.edu.ORCID 0000-0001-7555-8613
Rebekah L HudockDivision of Clinical Behavioral Neuroscience, Department of Pediatrics, University of Minnesota, 420 Delaware Street SE, Mayo Mail Code 486, Minneapolis, MN, 55455, USA.
Allison M FoyDivision of Clinical Behavioral Neuroscience, Department of Pediatrics, University of Minnesota, 420 Delaware Street SE, Mayo Mail Code 486, Minneapolis, MN, 55455, USA.
Margaret Semrud-ClikemanDivision of Clinical Behavioral Neuroscience, Department of Pediatrics, University of Minnesota, 420 Delaware Street SE, Mayo Mail Code 486, Minneapolis, MN, 55455, USA.
Mary Ella PierpontDivision of Genetics & Metabolism, Department of Pediatrics and Ophthalmology, University of Minnesota, 2450 Riverside Avenue, Minneapolis, MN, 55455, USA.
Susan A BerryDivision of Genetics & Metabolism, Department of Pediatrics and Ophthalmology, University of Minnesota, 2450 Riverside Avenue, Minneapolis, MN, 55455, USA.
Ryan ShanleyBiostatistics Core, University of Minnesota, 717 Delaware Street SE, Minneapolis, MN, 55414, USA.
Nathan RubinBiostatistics Core, University of Minnesota, 717 Delaware Street SE, Minneapolis, MN, 55414, USA.
Katherine SommerUniversity of Minnesota Health, 2450 Riverside Avenue, Minneapolis, MN, 55455, USA.
Christopher L MoertelDivision of Pediatric Hematology and Oncology, Department of Pediatrics, University of Minnesota, Mayo Mail Code 484, 420 Delaware Street SE, Mayo Mail Code 486, Minneapolis, MN, 55455, USA.
University of Minnesota · US

Funding

Women's CancerP30CA077598 · NCI · UNIVERSITY OF MINNESOTA TWIN CITIES · PI Douglas Yee · 1998 to 2026
$100.4M
University of Minnesota Clinical and Translational Science Institute (UMN CTSI)UL1TR000114 · NCATS · UNIVERSITY OF MINNESOTA · PI BLAZAR, BRUCE R · 2012 to 2015
$35.0M
University of Minnesota Clinical and Translational Science Institute (UMN CTSI)UL1TR002494 · NCATS · UNIVERSITY OF MINNESOTA · PI BLAZAR, BRUCE R, WEISDORF, DANIEL J · 2018 to 2022
$34.9M
NCATS NIH HHS UL1 TR000114NCATS NIH HHS UL1 TR002494NCI NIH HHS P30 CA077598
6 · The paper itself

Abstract

backgroundGene mutations within the RAS-MAPK signaling cascade result in Noonan syndrome (NS), neurofibromatosis type 1 (NF1), and related disorders. Recent research has documented an increased risk for social difficulties and features of autism spectrum disorder (ASD) among children with these conditions. Despite this emerging evidence, the neuropsychological characteristics associated with social skills deficits are not well understood, particularly for children with NS.

methodsParents of children with NS (n = 39), NF1 (n = 39), and unaffected siblings (n = 32) between the ages of 8 and 16 years were administered well-validated caregiver questionnaires assessing their child's social skills, language abilities, attention-deficit hyperactivity disorder (ADHD) symptoms and anxiety.

resultsWith respect to overall social skills, average ratings of children in both clinical groups were similar, and indicated weaker social skills compared to unaffected siblings. Although ratings of social skills were outside of normal limits for more than four in ten children within the clinical groups, most of the deficits were mild/moderate. Fifteen percent of the children with NS and 5% of the children with NF1 were rated as having severe social skills impairment (< - 2SD). Independent of diagnosis, having fewer ADHD symptoms or better social-pragmatic language skills was predictive of stronger social skills.

conclusionsAmidst efforts to support social skill development among children and adolescents with RASopathies, neuropsychological correlates such as social language competence, attention, and behavioral self-regulation could be important targets of intervention.

Indexed as

Social SkillsAdolescentChildFemaleHumansMaleNeurofibromatosis 1Neuropsychological TestsNoonan SyndromeLanguageNeurofibromatosis type 1NeuropsychologicalNF1Noonan syndromeRASopathiesSocial

Identifiers

PMID29914349
PMCPMC6006579
OpenAlexW2808175542

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.