Literature DB >> 22511892

Loss-of-Function CNKSR2 Mutation Is a Likely Cause of Non-Syndromic X-Linked Intellectual Disability.

G Houge1, I H Rasmussen, R Hovland.   

Abstract

In a non-dysmorphic 5-year-old boy with developmental delay, well-controlled epilepsy, and microcephaly, a 234-kb deletion of Xp22.12 was detected by copy number analysis. The maternally inherited deletion removed the initial 15 of the 21 exons of the connector enhancer of KSR-2 gene called CNKSR2 or CNK2. Our finding suggests that loss of CNKSR2 is a novel cause of non-syndromic X-linked mental retardation, an assumption supported by high gene expression in the brain, localization to the post-synaptic density, and a role in RAS/MAPK-dependent signal transduction.

Entities:  

Year:  2011        PMID: 22511892      PMCID: PMC3326275          DOI: 10.1159/000335159

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


  14 in total

1.  Densin-180, a synaptic protein, links to PSD-95 through its direct interaction with MAGUIN-1.

Authors:  Kazuhiro Ohtakara; Miwako Nishizawa; Ichiro Izawa; Yutaka Hata; Satoshi Matsushima; Waro Taki; Hiroyasu Inada; Yoshimi Takai; Masaki Inagaki
Journal:  Genes Cells       Date:  2002-11       Impact factor: 1.891

2.  CNK2 couples NGF signal propagation to multiple regulatory cascades driving cell differentiation.

Authors:  Ron Bumeister; Carine Rosse; Anthony Anselmo; Jacques Camonis; Michael A White
Journal:  Curr Biol       Date:  2004-03-09       Impact factor: 10.834

3.  Prediction of the coding sequences of unidentified human genes. XII. The complete sequences of 100 new cDNA clones from brain which code for large proteins in vitro.

Authors:  T Nagase; K Ishikawa; M Suyama; R Kikuno; M Hirosawa; N Miyajima; A Tanaka; H Kotani; N Nomura; O Ohara
Journal:  DNA Res       Date:  1998-12-31       Impact factor: 4.458

4.  Proteomic characterization of the dynamic KSR-2 interactome, a signaling scaffold complex in MAPK pathway.

Authors:  Lin Liu; Padma L Channavajhala; Vikram R Rao; Ioannis Moutsatsos; Leeying Wu; Yuhua Zhang; Lih-Ling Lin; Yongchang Qiu
Journal:  Biochim Biophys Acta       Date:  2009-06-27

5.  A Raf-induced allosteric transition of KSR stimulates phosphorylation of MEK.

Authors:  Damian F Brennan; Arvin C Dar; Nicholas T Hertz; William C H Chao; Alma L Burlingame; Kevan M Shokat; David Barford
Journal:  Nature       Date:  2011-03-27       Impact factor: 49.962

6.  MAGUIN, a novel neuronal membrane-associated guanylate kinase-interacting protein.

Authors:  I Yao; Y Hata; N Ide; K Hirao; M Deguchi; H Nishioka; A Mizoguchi; Y Takai
Journal:  J Biol Chem       Date:  1999-04-23       Impact factor: 5.157

7.  A KSR/CNK complex mediated by HYP, a novel SAM domain-containing protein, regulates RAS-dependent RAF activation in Drosophila.

Authors:  Mélanie Douziech; Malha Sahmi; Gino Laberge; Marc Therrien
Journal:  Genes Dev       Date:  2006-04-01       Impact factor: 11.361

8.  KSR is a scaffold required for activation of the ERK/MAPK module.

Authors:  François Roy; Gino Laberge; Mélanie Douziech; David Ferland-McCollough; Marc Therrien
Journal:  Genes Dev       Date:  2002-02-15       Impact factor: 11.361

9.  Synaptic localization of membrane-associated guanylate kinase-interacting protein mediated by the pleckstrin homology domain.

Authors:  Junko Iida; Wataru Nishimura; Ikuko Yao; Yutaka Hata
Journal:  Eur J Neurosci       Date:  2002-05       Impact factor: 3.386

10.  CNK, a RAF-binding multidomain protein required for RAS signaling.

Authors:  M Therrien; A M Wong; G M Rubin
Journal:  Cell       Date:  1998-10-30       Impact factor: 41.582

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  22 in total

1.  XLID-causing mutations and associated genes challenged in light of data from large-scale human exome sequencing.

Authors:  Amélie Piton; Claire Redin; Jean-Louis Mandel
Journal:  Am J Hum Genet       Date:  2013-07-18       Impact factor: 11.025

2.  Molecular architecture of postsynaptic Interactomes.

Authors:  Brent Wilkinson; Marcelo P Coba
Journal:  Cell Signal       Date:  2020-09-14       Impact factor: 4.315

3.  Recurrent De Novo Mutations Disturbing the GTP/GDP Binding Pocket of RAB11B Cause Intellectual Disability and a Distinctive Brain Phenotype.

Authors:  Ideke J C Lamers; Margot R F Reijnders; Hanka Venselaar; Alison Kraus; Sandra Jansen; Bert B A de Vries; Gunnar Houge; Gyri Aasland Gradek; Jieun Seo; Murim Choi; Jong-Hee Chae; Ineke van der Burgt; Rolph Pfundt; Stef J F Letteboer; Sylvia E C van Beersum; Simone Dusseljee; Han G Brunner; Dan Doherty; Tjitske Kleefstra; Ronald Roepman
Journal:  Am J Hum Genet       Date:  2017-10-26       Impact factor: 11.025

4.  Experience-dependent neuroplasticity of the developing hypothalamus: integrative epigenomic approaches.

Authors:  Annie Vogel Ciernia; Benjamin I Laufer; Keith W Dunaway; Charles E Mordaunt; Rochelle L Coulson; Theresa S Totah; Danielle S Stolzenberg; Jaime C Frahm; Akanksha Singh-Taylor; Tallie Z Baram; Janine M LaSalle; Dag H Yasui
Journal:  Epigenetics       Date:  2018-05-10       Impact factor: 4.528

5.  De novo variants in SETD1B are associated with intellectual disability, epilepsy and autism.

Authors:  Takuya Hiraide; Mitsuko Nakashima; Kaori Yamoto; Tokiko Fukuda; Mitsuhiro Kato; Hiroko Ikeda; Yoko Sugie; Kazushi Aoto; Tadashi Kaname; Kazuhiko Nakabayashi; Tsutomu Ogata; Naomichi Matsumoto; Hirotomo Saitsu
Journal:  Hum Genet       Date:  2018-01-10       Impact factor: 4.132

6.  Brain-specific deletion of GIT1 impairs cognition and alters phosphorylation of synaptic protein networks implicated in schizophrenia susceptibility.

Authors:  Daniel M Fass; Michael C Lewis; Rushdy Ahmad; Matthew J Szucs; Qiangge Zhang; Morgan Fleishman; Dongqing Wang; Myung Jong Kim; Jonathan Biag; Steven A Carr; Edward M Scolnick; Richard T Premont; Stephen J Haggarty
Journal:  Mol Psychiatry       Date:  2022-05-03       Impact factor: 15.992

7.  The CNK2 scaffold interacts with vilse and modulates Rac cycling during spine morphogenesis in hippocampal neurons.

Authors:  Junghwa Lim; Daniel A Ritt; Ming Zhou; Deborah K Morrison
Journal:  Curr Biol       Date:  2014-03-20       Impact factor: 10.834

8.  MECP2 Is a Frequently Amplified Oncogene with a Novel Epigenetic Mechanism That Mimics the Role of Activated RAS in Malignancy.

Authors:  Manish Neupane; Allison P Clark; Serena Landini; Nicolai J Birkbak; Aron C Eklund; Elgene Lim; Aedin C Culhane; William T Barry; Steven E Schumacher; Rameen Beroukhim; Zoltan Szallasi; Marc Vidal; David E Hill; Daniel P Silver
Journal:  Cancer Discov       Date:  2015-11-06       Impact factor: 39.397

9.  Cnksr2 Loss in Mice Leads to Increased Neural Activity and Behavioral Phenotypes of Epilepsy-Aphasia Syndrome.

Authors:  Eda Erata; Yudong Gao; Alicia M Purkey; Erik J Soderblom; James O McNamara; Scott H Soderling
Journal:  J Neurosci       Date:  2021-09-27       Impact factor: 6.167

10.  CNKSR2 gene mutation leads to Houge type of X-linked syndromic mental retardation: A case report and review of literature.

Authors:  Qingyun Kang; Liming Yang; Hongmei Liao; Liwen Wu; Bo Chen; Sai Yang; Xiaojun Kuang; Haiyang Yang; Caishi Liao
Journal:  Medicine (Baltimore)       Date:  2021-06-11       Impact factor: 1.817

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