Literature DB >> 33510598

Language Impairment with a Partial Duplication of DOCK8.

Antonio Benítez-Burraco1, Maite Fernández-Urquiza2, Mª Salud Jiménez-Romero3.   

Abstract

Duplications of the distal region of the short arm of chromosome 9 are rare, but are associated with learning disabilities and behavioral disturbances. We report in detail the cognitive and language features of a child with a duplication in the 9p24.3 region, arr[hg19] 9p24.3(266,045-459,076)×3. The proband exhibits marked expressive and receptive problems, which affect both structural and functional aspects of language. These problems might result from a severe underlying deficit in working memory. Regarding the molecular causes of the observed symptoms, they might result from the altered expression of selected genes involved in procedural learning, particularly some of components of the SLIT/ROBO/FOXP2 network, strongly related to the development and evolution of language. Dysregulation of specific components of this network can result in turn from an altered interaction between DOCK8, affected by the microduplication, and CDC42, acting as the hub component of the network encompassing language-related genes.
Copyright © 2020 by S. Karger AG, Basel.

Entities:  

Keywords:  9p24.3 duplication; Cognitive delay; DOCK8; Language deficits; SLIT/ROBO/FOXP2; Working memory

Year:  2020        PMID: 33510598      PMCID: PMC7802463          DOI: 10.1159/000511972

Source DB:  PubMed          Journal:  Mol Syndromol        ISSN: 1661-8769


  82 in total

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Review 2.  Dynamic FoxO transcription factors.

Authors:  Haojie Huang; Donald J Tindall
Journal:  J Cell Sci       Date:  2007-08-01       Impact factor: 5.285

3.  Functional investigation of a schizophrenia GWAS signal at the CDC42 gene.

Authors:  William P Gilks; Matthew Hill; Michael Gill; Gary Donohoe; Aiden P Corvin; Derek W Morris
Journal:  World J Biol Psychiatry       Date:  2012-03-05       Impact factor: 4.132

Review 4.  DOCK8 deficiency: Insights into pathophysiology, clinical features and management.

Authors:  Catherine M Biggs; Sevgi Keles; Talal A Chatila
Journal:  Clin Immunol       Date:  2017-06-15       Impact factor: 3.969

5.  Focal adhesion kinase modulates Cdc42 activity downstream of positive and negative axon guidance cues.

Authors:  Jonathan P Myers; Estuardo Robles; Allison Ducharme-Smith; Timothy M Gomez
Journal:  J Cell Sci       Date:  2012-03-05       Impact factor: 5.285

6.  Neuronal cell adhesion molecule deletion induces a cognitive and behavioral phenotype reflective of impulsivity.

Authors:  L D Matzel; J Babiarz; D A Townsend; H C Grossman; M Grumet
Journal:  Genes Brain Behav       Date:  2007-12-06       Impact factor: 3.449

7.  Globularity and language-readiness: generating new predictions by expanding the set of genes of interest.

Authors:  Cedric Boeckx; Antonio Benítez-Burraco
Journal:  Front Psychol       Date:  2014-11-25

8.  Further evidence for a parent-of-origin effect at the NOP9 locus on language-related phenotypes.

Authors:  Kerry A Pettigrew; Emily Frinton; Ron Nudel; May T M Chan; Paul Thompson; Marianna E Hayiou-Thomas; Joel B Talcott; John Stein; Anthony P Monaco; Charles Hulme; Margaret J Snowling; Dianne F Newbury; Silvia Paracchini
Journal:  J Neurodev Disord       Date:  2016-06-14       Impact factor: 4.025

9.  Expression and Function of Neuron-Glia-Related Cell Adhesion Molecule (NrCAM) in the Amygdalar Pathway.

Authors:  Vishwa Mohan; Julia R Gomez; Patricia F Maness
Journal:  Front Cell Dev Biol       Date:  2019-01-31

10.  The Oscillopathic Nature of Language Deficits in Autism: From Genes to Language Evolution.

Authors:  Antonio Benítez-Burraco; Elliot Murphy
Journal:  Front Hum Neurosci       Date:  2016-03-18       Impact factor: 3.169

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  1 in total

1.  Identification of copy number variants in children and adolescents with autism spectrum disorder: a study from Turkey.

Authors:  Ahmet Özaslan; Gülsüm Kayhan; Elvan İşeri; Mehmet Ali Ergün; Esra Güney; Ferda Emriye Perçin
Journal:  Mol Biol Rep       Date:  2021-10-12       Impact factor: 2.316

  1 in total

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