Literature DB >> 25388972

Prevalence of Autism Spectrum Disorder symptoms in children with neurofibromatosis type 1.

Ellen Plasschaert1, Mie-Jef Descheemaeker, Lien Van Eylen, Ilse Noens, Jean Steyaert, Eric Legius.   

Abstract

Neurofibromatosis type 1 (NF1) is an autosomal dominant genetic condition. While considerable work has focused on cognitive functioning, several research groups also observed difficulties in social functioning as a prominent feature of NF1. These problems and the possible link between NF1 and Autism Spectrum Disorder (ASD) have become increasingly important in recent NF1 literature. The aim of the current study was to assess ASD characteristics in a hospital-based NF1 pediatric population (n = 82) using the standardized Children Social Behavior Questionnaire (CSBQ) and Social Responsiveness Scale (SRS) to account for the prevalence, severity, and nature of social problems. In a parallel study, comprehensive ASD assessment was performed in a subgroup of NF1 children with a strong suspicion of ASD (n = 31). Results indicate that NF1 children have more social problems than typical controls, more frequently reported above 8 years. The SRS shows that 63% is at risk of ASD symptoms. According to item analyses, most problems were observed on items measuring orientation in, understanding of and being tuned onto a social situation (CSBQ) and social cognition and communication (SRS). In the parallel study, 27 NF1 children were diagnosed with ASD. These children have a distinct phenotype compared to a heterogeneous ASD group, with pronounced social-communicative impairments and fewer restrictive/repetitive behaviors. This study provides a better understanding of social problems in NF1 and the phenotypical overlap with ASD symptomatology. Despite their willingness to engage with others, NF1 children with or without ASD encounter various difficulties in their social-communicative life.
© 2014 Wiley Periodicals, Inc.

Entities:  

Keywords:  ASD; NF1; children social behavior questionnaire; social; social responsiveness scale

Mesh:

Year:  2014        PMID: 25388972     DOI: 10.1002/ajmg.b.32280

Source DB:  PubMed          Journal:  Am J Med Genet B Neuropsychiatr Genet        ISSN: 1552-4841            Impact factor:   3.568


  36 in total

1.  RASopathies are associated with a distinct personality profile.

Authors:  Varoona Bizaoui; Jessica Gage; Rita Brar; Katherine A Rauen; Lauren A Weiss
Journal:  Am J Med Genet B Neuropsychiatr Genet       Date:  2018-04-16       Impact factor: 3.568

2.  Identifying Symptoms of Distress in Youth Living with Neurofibromatosis Type 1 (NF1).

Authors:  Lori Wiener; Haven Battles; Sima Zadeh Bedoya; Andrea Baldwin; Brigitte C Widemann; Maryland Pao
Journal:  J Genet Couns       Date:  2017-07-23       Impact factor: 2.537

3.  Behavioral and cognitive outcomes for clinical trials in children with neurofibromatosis type 1.

Authors:  Thijs van der Vaart; André B Rietman; Ellen Plasschaert; Eric Legius; Ype Elgersma; Henriëtte A Moll
Journal:  Neurology       Date:  2015-10-30       Impact factor: 9.910

4.  Social Functioning and Behaviour in Mucopolysaccharidosis IH [Hurlers Syndrome].

Authors:  Annukka Lehtonen; Stewart Rust; Simon Jones; Richard Brown; Dougal Hare
Journal:  JIMD Rep       Date:  2017-07-29

5.  Social Function and Autism Spectrum Disorder in Children and Adults with Neurofibromatosis Type 1: a Systematic Review and Meta-Analysis.

Authors:  Anita K Chisholm; Vicki A Anderson; Natalie A Pride; Stephanie Malarbi; Kathryn N North; Jonathan M Payne
Journal:  Neuropsychol Rev       Date:  2018-08-11       Impact factor: 7.444

6.  Distribution and Within-Family Specificity of Quantitative Autistic Traits in Patients with Neurofibromatosis Type I.

Authors:  John N Constantino; Yi Zhang; Kieran Holzhauer; Sayli Sant; Kyna Long; Alicia Vallorani; Leena Malik; David H Gutmann
Journal:  J Pediatr       Date:  2015-06-04       Impact factor: 4.406

7.  Social Behavioral Deficits with Loss of Neurofibromin Emerge from Peripheral Chemosensory Neuron Dysfunction.

Authors:  Emilia H Moscato; Christine Dubowy; James A Walker; Matthew S Kayser
Journal:  Cell Rep       Date:  2020-07-07       Impact factor: 9.423

Review 8.  Discovery of Rare Mutations in Autism: Elucidating Neurodevelopmental Mechanisms.

Authors:  Ece D Gamsiz; Laura N Sciarra; Abbie M Maguire; Matthew F Pescosolido; Laura I van Dyck; Eric M Morrow
Journal:  Neurotherapeutics       Date:  2015-07       Impact factor: 7.620

9.  An Internet support group for parents of children with neurofibromatosis type 1: a qualitative analysis.

Authors:  Staci Martin; Kari L Struemph; Alyssa Poblete; Mary Anne Toledo-Tamula; Robin Lockridge; Marie Claire Roderick; Pamela Wolters
Journal:  J Community Genet       Date:  2018-03-02

10.  Brief Report: The Prevalence of Neurofibromatosis Type 1 among Children with Autism Spectrum Disorder Identified by the Autism and Developmental Disabilities Monitoring Network.

Authors:  Deborah A Bilder; Amanda V Bakian; David A Stevenson; Paul S Carbone; Christopher Cunniff; Alyson B Goodman; William M McMahon; Nicole P Fisher; David Viskochil
Journal:  J Autism Dev Disord       Date:  2016-10
View more

北京卡尤迪生物科技股份有限公司 © 2022-2023.