Literature DB >> 23885228

Towards identification of individual etiologies by resolving genomic and biological conundrums in patients with autism spectrum disorders.

M Poot1.   

Abstract

Recent genomic research into autism spectrum disorders (ASD) has revealed a remarkably complex genetic architecture. Large numbers of common variants, copy number variations and single nucleotide variants have been identified, yet each of them individually afforded only a small phenotypic impact. A polygenic model in which multiple genes interact either in an additive or a synergistic way appears the most plausible for the majority of ASD patients. Based on recently identified ASD candidate genes, transgenic mouse models for neuroligins/neurorexins and genes such as Cntnap2, Cntn5, Tsc1, Tsc2, Akt3, Cyfip1, Scn1a, En2, Slc6a4, and Bckdk have been generated and studied with respect to behavioral and neuroanatomical phenotypes and sensitivity to drug treatments. From these models, a few clues for potential pharmacologic intervention emerged. The Fmr1, Shank2 and Cntn5 knockout mice exhibited alterations of glutamate receptors, which may become a target for pharmacologic modulation. Some of the phenotypes of Mecp2 knockout mice can be ameliorated by administering IGF1. In the near future, comprehensive genotyping of individual patients and siblings combined with the novel insights generated from the transgenic animal studies may provide us with personalized treatment options. Eventually, autism may indeed turn out to be a phenotypically heterogeneous group of disorders ('autisms') caused by combinations of changes in multiple possible candidate genes, being different in each patient and requiring for each combination of mutations a distinct, individually tailored treatment.

Entities:  

Keywords:  Autism; Copy number variation; Personalized medicine; Polygenic mode; Single nucleotide variation; Transgenic mouse models

Year:  2013        PMID: 23885228      PMCID: PMC3711481          DOI: 10.1159/000350041

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


  85 in total

Review 1.  Animal models of neuropsychiatric disorders.

Authors:  Eric J Nestler; Steven E Hyman
Journal:  Nat Neurosci       Date:  2010-09-27       Impact factor: 24.884

2.  An autism-associated point mutation in the neuroligin cytoplasmic tail selectively impairs AMPA receptor-mediated synaptic transmission in hippocampus.

Authors:  Mark R Etherton; Katsuhiko Tabuchi; Manu Sharma; Jaewon Ko; Thomas C Südhof
Journal:  EMBO J       Date:  2011-06-03       Impact factor: 11.598

Review 3.  Genome arrays for the detection of copy number variations in idiopathic mental retardation, idiopathic generalized epilepsy and neuropsychiatric disorders: lessons for diagnostic workflow and research.

Authors:  R Hochstenbach; J E Buizer-Voskamp; J A S Vorstman; R A Ophoff
Journal:  Cytogenet Genome Res       Date:  2011-11-02       Impact factor: 1.636

Review 4.  Genes associated with autism spectrum disorder.

Authors:  Xiaohong Li; Hua Zou; W Ted Brown
Journal:  Brain Res Bull       Date:  2012-06-09       Impact factor: 4.077

Review 5.  Phenotypic variability and genetic susceptibility to genomic disorders.

Authors:  Santhosh Girirajan; Evan E Eichler
Journal:  Hum Mol Genet       Date:  2010-08-31       Impact factor: 6.150

Review 6.  A review on the current neuroligin mouse models.

Authors:  Jun-Yu Xu; Qiang-Qiang Xia; Jun Xia
Journal:  Sheng Li Xue Bao       Date:  2012-10-25

7.  Autism genome-wide copy number variation reveals ubiquitin and neuronal genes.

Authors:  Joseph T Glessner; Kai Wang; Guiqing Cai; Olena Korvatska; Cecilia E Kim; Shawn Wood; Haitao Zhang; Annette Estes; Camille W Brune; Jonathan P Bradfield; Marcin Imielinski; Edward C Frackelton; Jennifer Reichert; Emily L Crawford; Jeffrey Munson; Patrick M A Sleiman; Rosetta Chiavacci; Kiran Annaiah; Kelly Thomas; Cuiping Hou; Wendy Glaberson; James Flory; Frederick Otieno; Maria Garris; Latha Soorya; Lambertus Klei; Joseph Piven; Kacie J Meyer; Evdokia Anagnostou; Takeshi Sakurai; Rachel M Game; Danielle S Rudd; Danielle Zurawiecki; Christopher J McDougle; Lea K Davis; Judith Miller; David J Posey; Shana Michaels; Alexander Kolevzon; Jeremy M Silverman; Raphael Bernier; Susan E Levy; Robert T Schultz; Geraldine Dawson; Thomas Owley; William M McMahon; Thomas H Wassink; John A Sweeney; John I Nurnberger; Hilary Coon; James S Sutcliffe; Nancy J Minshew; Struan F A Grant; Maja Bucan; Edwin H Cook; Joseph D Buxbaum; Bernie Devlin; Gerard D Schellenberg; Hakon Hakonarson
Journal:  Nature       Date:  2009-04-28       Impact factor: 49.962

8.  Partial reversal of Rett Syndrome-like symptoms in MeCP2 mutant mice.

Authors:  Daniela Tropea; Emanuela Giacometti; Nathan R Wilson; Caroline Beard; Cortina McCurry; Dong Dong Fu; Ruth Flannery; Rudolf Jaenisch; Mriganka Sur
Journal:  Proc Natl Acad Sci U S A       Date:  2009-02-10       Impact factor: 11.205

9.  Strong association of de novo copy number mutations with autism.

Authors:  Jonathan Sebat; B Lakshmi; Dheeraj Malhotra; Jennifer Troge; Christa Lese-Martin; Tom Walsh; Boris Yamrom; Seungtai Yoon; Alex Krasnitz; Jude Kendall; Anthony Leotta; Deepa Pai; Ray Zhang; Yoon-Ha Lee; James Hicks; Sarah J Spence; Annette T Lee; Kaija Puura; Terho Lehtimäki; David Ledbetter; Peter K Gregersen; Joel Bregman; James S Sutcliffe; Vaidehi Jobanputra; Wendy Chung; Dorothy Warburton; Mary-Claire King; David Skuse; Daniel H Geschwind; T Conrad Gilliam; Kenny Ye; Michael Wigler
Journal:  Science       Date:  2007-03-15       Impact factor: 47.728

Review 10.  Autism and medical disorders: a review of the literature.

Authors:  C Gillberg; M Coleman
Journal:  Dev Med Child Neurol       Date:  1996-03       Impact factor: 5.449

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  11 in total

1.  SHANK Mutations May Disorder Brain Development.

Authors:  Martin Poot
Journal:  Mol Syndromol       Date:  2015-02

2.  Late breaking chromosomes.

Authors:  M Poot
Journal:  Mol Syndromol       Date:  2013-06

3.  The Age of the Father.

Authors:  Martin Poot
Journal:  Mol Syndromol       Date:  2017-04-20

4.  Syndromes Hidden within the 16p11.2 Deletion Region.

Authors:  Martin Poot
Journal:  Mol Syndromol       Date:  2018-07-13

5.  Late breaking chromosomes.

Authors:  M Poot
Journal:  Mol Syndromol       Date:  2014-01

Review 6.  Connecting the CNTNAP2 Networks with Neurodevelopmental Disorders.

Authors:  Martin Poot
Journal:  Mol Syndromol       Date:  2015-02-03

7.  A candidate gene association study further corroborates involvement of contactin genes in autism.

Authors:  Martin Poot
Journal:  Mol Syndromol       Date:  2014-05-17

8.  Microdeletions including FMR1 in three female patients with intellectual disability - further delineation of the phenotype and expression studies.

Authors:  A M Zink; E Wohlleber; H Engels; O K Rødningen; K Ravn; S Heilmann; J Rehnitz; N Katzorke; C Kraus; S Blichfeldt; P Hoffmann; H Reutter; F F Brockschmidt; M Kreiß-Nachtsheim; P H Vogt; T E Prescott; Z Tümer; J A Lee
Journal:  Mol Syndromol       Date:  2014-01-29

Review 9.  Mechanisms of Origin, Phenotypic Effects and Diagnostic Implications of Complex Chromosome Rearrangements.

Authors:  Martin Poot; Thomas Haaf
Journal:  Mol Syndromol       Date:  2015-08-15

10.  The GRIA3 c.2477G > A Variant Causes an Exaggerated Startle Reflex, Chorea, and Multifocal Myoclonus.

Authors:  Juliette Piard; Matthieu Béreau; Wenshu XiangWei; Thomas Wirth; Daniel Amsallem; Lauren Buisson; Philippe Richard; Nana Liu; Yuchen Xu; Scott J Myers; Stephen F Traynelis; Jameleddine Chelly; Mathieu Anheim; Martine Raynaud; Lionel Van Maldergem; Hongjie Yuan
Journal:  Mov Disord       Date:  2020-05-05       Impact factor: 9.698

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