Literature DB >> 22986108

A 1 Mb de novo deletion within 11q13.1q13.2 in a boy with mild intellectual disability and minor dysmorphic features.

Karijn Floor1, Tuva Barøy, Doriana Misceo, Oivind J Kanavin, Madeleine Fannemel, Eirik Frengen.   

Abstract

We report a 11 year old male patient ascertained for mild intellectual disability and minor dysmorphic features, carrying a 1 Mb de novo deletion on chromosome 11q13.1q13.2 detected by aCGH. This is the first report of a deletion in this region in a patient presenting with intellectual impairment and mild dysmorphic traits. The 1 Mb deleted area encompasses 47 RefSeq genes, including Cornichon homologue 2 (CNIH2), Cofilin-1 (CFL1) and neuronal PAS domain-containing protein 4 (NPAS4), which are highly expressed in the central nervous system. Knockout of the CNIH2 and CFL1 orthologues in animals results in migration disturbances, while low or no expression of Npas4 in mice results in impairment of memory and learning. These three genes have previously been suggested as candidate genes for neurological disorders.
Copyright © 2012 Elsevier Masson SAS. All rights reserved.

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Year:  2012        PMID: 22986108     DOI: 10.1016/j.ejmg.2012.08.002

Source DB:  PubMed          Journal:  Eur J Med Genet        ISSN: 1769-7212            Impact factor:   2.708


  8 in total

1.  Sex Differences in the Role of CNIH3 on Spatial Memory and Synaptic Plasticity.

Authors:  Hannah E Frye; Yukitoshi Izumi; Alexis N Harris; Sidney B Williams; Christopher R Trousdale; Min-Yu Sun; Andrew D Sauerbeck; Terrance T Kummer; Steven Mennerick; Charles F Zorumski; Elliot C Nelson; Joseph D Dougherty; Jose A Morón
Journal:  Biol Psychiatry       Date:  2021-07-23       Impact factor: 13.382

Review 2.  Glutamate receptor mutations in psychiatric and neurodevelopmental disorders.

Authors:  David Soto; Xavier Altafaj; Carlos Sindreu; Alex Bayés
Journal:  Commun Integr Biol       Date:  2014-02-06

Review 3.  The Role of the Neuroprotective Factor Npas4 in Cerebral Ischemia.

Authors:  Fong Chan Choy; Thomas S Klarić; Simon A Koblar; Martin D Lewis
Journal:  Int J Mol Sci       Date:  2015-12-04       Impact factor: 5.923

4.  Molecular characterisation of rare loss-of-function NPAS3 and NPAS4 variants identified in individuals with neurodevelopmental disorders.

Authors:  Joseph J Rossi; Jill A Rosenfeld; Katie M Chan; Haley Streff; Victoria Nankivell; Daniel J Peet; Murray L Whitelaw; David C Bersten
Journal:  Sci Rep       Date:  2021-03-23       Impact factor: 4.996

Review 5.  AMPA receptor structure and auxiliary subunits.

Authors:  Aichurok Kamalova; Terunaga Nakagawa
Journal:  J Physiol       Date:  2020-02-18       Impact factor: 5.182

6.  Human variants in the neuronal basic helix-loop-helix/Per-Arnt-Sim (bHLH/PAS) transcription factor complex NPAS4/ARNT2 disrupt function.

Authors:  David C Bersten; John B Bruning; Daniel J Peet; Murray L Whitelaw
Journal:  PLoS One       Date:  2014-01-17       Impact factor: 3.240

7.  AMPA Receptor Auxiliary Subunit GSG1L Suppresses Short-Term Facilitation in Corticothalamic Synapses and Determines Seizure Susceptibility.

Authors:  Aichurok Kamalova; Kensuke Futai; Eric Delpire; Terunaga Nakagawa
Journal:  Cell Rep       Date:  2020-07-21       Impact factor: 9.423

8.  The Bdnf and Npas4 genes are targets of HDAC3-mediated transcriptional repression.

Authors:  Anto Sam Crosslee Louis Sam Titus; Dharmendra Sharma; Min Soo Kim; Santosh R D'Mello
Journal:  BMC Neurosci       Date:  2019-12-28       Impact factor: 3.288

  8 in total

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