Literature DB >> 33562463

Whole Exome Sequencing Reveals a Novel AUTS2 In-Frame Deletion in a Boy with Global Developmental Delay, Absent Speech, Dysmorphic Features, and Cerebral Anomalies.

Pietro Palumbo1, Ester Di Muro1, Maria Accadia2, Mario Benvenuto1, Marilena Carmela Di Giacomo3, Stefano Castellana4, Tommaso Mazza4, Marco Castori1, Orazio Palumbo1, Massimo Carella1.   

Abstract

Neurodevelopmental disorders (NDDs) are a group of highly prevalent, clinically and genetically heterogeneous pediatric disorders comprising, according to the Diagnostic and Statistical Manual of Mental Disorders 5th edition (DSM-V), intellectual disability, developmental delay, autism spectrum disorders, and other neurological and cognitive disorders manifesting in the developmental age. To date, more than 1000 genes have been implicated in the etiopathogenesis of NNDs. Among them, AUTS2 (OMIM # 607270) encodes a protein involved in neural migration and neuritogenesis, and causes NNDs with different molecular mechanisms including copy number variations, single or multiple exonic deletion and single nucleotide variants. We describes a 9-year-old boy with global developmental delay, absent speech, minor craniofacial anomalies, hypoplasia of the cerebellar vermis and thinning of the corpus callosum, resulted carrier of the de novo AUTS2 c.1603_1626del deletion at whole exome sequencing (WES) predicted to cause the loss of eight amino acids [p.(His535_Thr542del)]. Notably, our patient is the first reported so far in medical literature carrying an in-frame deletion and the first in which absent language, hypoplasia of the cerebellar vermis and thinning of the corpus callosum has been observed thus useful to expand the molecular spectrum of AUTS2 pathogenic variants and to broaden our knowledge on the clinical phenotype associated.

Entities:  

Keywords:  AUTS2; neurodevelopmental disorders; whole exome sequencing

Mesh:

Substances:

Year:  2021        PMID: 33562463      PMCID: PMC7915150          DOI: 10.3390/genes12020229

Source DB:  PubMed          Journal:  Genes (Basel)        ISSN: 2073-4425            Impact factor:   4.096


  42 in total

Review 1.  The genetics of neurodevelopmental disease.

Authors:  Kevin J Mitchell
Journal:  Curr Opin Neurobiol       Date:  2010-09-09       Impact factor: 6.627

2.  TBR1 is the candidate gene for intellectual disability in patients with a 2q24.2 interstitial deletion.

Authors:  Orazio Palumbo; Marco Fichera; Pietro Palumbo; Renata Rizzo; Elisabetta Mazzolla; Donatella Maria Cocuzza; Massimo Carella; Teresa Mattina
Journal:  Am J Med Genet A       Date:  2014-01-23       Impact factor: 2.802

3.  A de novo balanced translocation breakpoint truncating the autism susceptibility candidate 2 (AUTS2) gene in a patient with autism.

Authors:  Xin-Li Huang; Ying S Zou; Tom A Maher; Stephanie Newton; Jeff M Milunsky
Journal:  Am J Med Genet A       Date:  2010-08       Impact factor: 2.802

4.  Genome-wide copy number variation in epilepsy: novel susceptibility loci in idiopathic generalized and focal epilepsies.

Authors:  Heather C Mefford; Hiltrud Muhle; Philipp Ostertag; Sarah von Spiczak; Karen Buysse; Carl Baker; Andre Franke; Alain Malafosse; Pierre Genton; Pierre Thomas; Christina A Gurnett; Stefan Schreiber; Alexander G Bassuk; Michel Guipponi; Ulrich Stephani; Ingo Helbig; Evan E Eichler
Journal:  PLoS Genet       Date:  2010-05-20       Impact factor: 5.917

5.  Molecular cytogenetic analysis and resequencing of contactin associated protein-like 2 in autism spectrum disorders.

Authors:  Betul Bakkaloglu; Brian J O'Roak; Angeliki Louvi; Abha R Gupta; Jesse F Abelson; Thomas M Morgan; Katarzyna Chawarska; Ami Klin; A Gulhan Ercan-Sencicek; Althea A Stillman; Gamze Tanriover; Brett S Abrahams; Jackie A Duvall; Elissa M Robbins; Daniel H Geschwind; Thomas Biederer; Murat Gunel; Richard P Lifton; Matthew W State
Journal:  Am J Hum Genet       Date:  2008-01       Impact factor: 11.025

6.  GDF5 mutation case report and a systematic review of molecular and clinical spectrum: Expanding current knowledge on genotype-phenotype correlations.

Authors:  Maria Luce Genovesi; Daniele Guadagnolo; Enrica Marchionni; Agnese Giovannetti; Alice Traversa; Noemi Panzironi; Silvia Bernardo; Pietro Palumbo; Francesco Petrizzelli; Massimo Carella; Tommaso Mazza; Antonio Pizzuti; Viviana Caputo
Journal:  Bone       Date:  2021-01-12       Impact factor: 4.398

7.  Relative burden of large CNVs on a range of neurodevelopmental phenotypes.

Authors:  Santhosh Girirajan; Zoran Brkanac; Bradley P Coe; Carl Baker; Laura Vives; Tiffany H Vu; Neil Shafer; Raphael Bernier; Giovanni B Ferrero; Margherita Silengo; Stephen T Warren; Carlos S Moreno; Marco Fichera; Corrado Romano; Wendy H Raskind; Evan E Eichler
Journal:  PLoS Genet       Date:  2011-11-10       Impact factor: 5.917

8.  Genome-wide distribution of Auts2 binding localizes with active neurodevelopmental genes.

Authors:  N Oksenberg; G D E Haliburton; W L Eckalbar; I Oren; S Nishizaki; K Murphy; K S Pollard; R Y Birnbaum; N Ahituv
Journal:  Transl Psychiatry       Date:  2014-09-02       Impact factor: 6.222

9.  An AUTS2-Polycomb complex activates gene expression in the CNS.

Authors:  Zhonghua Gao; Pedro Lee; James M Stafford; Melanie von Schimmelmann; Anne Schaefer; Danny Reinberg
Journal:  Nature       Date:  2014-12-18       Impact factor: 49.962

Review 10.  Neuronal Migration and AUTS2 Syndrome.

Authors:  Kei Hori; Mikio Hoshino
Journal:  Brain Sci       Date:  2017-05-14
View more
  3 in total

Review 1.  AUTS2 Gene: Keys to Understanding the Pathogenesis of Neurodevelopmental Disorders.

Authors:  Kei Hori; Kazumi Shimaoka; Mikio Hoshino
Journal:  Cells       Date:  2021-12-21       Impact factor: 6.600

2.  Trio-based exome sequencing reveals a high rate of the de novo variants in intellectual disability.

Authors:  Alejandro J Brea-Fernández; Miriam Álvarez-Barona; Jorge Amigo; María Tubío-Fungueiriño; Pilar Caamaño; Montserrat Fernández-Prieto; Francisco Barros; Silvia De Rubeis; Joseph Buxbaum; Ángel Carracedo
Journal:  Eur J Hum Genet       Date:  2022-03-23       Impact factor: 5.351

Review 3.  Attention Deficit Hyperactivity and Autism Spectrum Disorders as the Core Symptoms of AUTS2 Syndrome: Description of Five New Patients and Update of the Frequency of Manifestations and Genotype-Phenotype Correlation.

Authors:  Carolina Sanchez-Jimeno; Fiona Blanco-Kelly; Fermina López-Grondona; Rebeca Losada-Del Pozo; Beatriz Moreno; María Rodrigo-Moreno; Elena Martinez-Cayuelas; Rosa Riveiro-Alvarez; María Fenollar-Cortés; Carmen Ayuso; Marta Rodríguez de Alba; Isabel Lorda-Sanchez; Berta Almoguera
Journal:  Genes (Basel)       Date:  2021-08-30       Impact factor: 4.096

  3 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.