Literature DB >> 15776425

Identification of ten novel mutations in patients with eIF2B-related disorders.

Andreas Ohlenbusch1, Marco Henneke, Knut Brockmann, Maria Goerg, Folker Hanefeld, Alfried Kohlschütter, Jutta Gärtner.   

Abstract

Autosomal recessive inherited mutations in each of the five eukaryotic initiation factor 2B (eIF2B) subunits are known to cause white matter abnormalities with a wide continuum of clinical signs and severity leading to the concept of eIF2B-related disorders. The clinical spectrum extends from fatal infantile forms to adult forms with slow or absent neurological deterioration. In this study 15 well-characterised patients with the classical form of leukoencephalopathy with vanishing white matter (VWM) or with phenotypic variants like ovarioleukodystrophy were investigated for mutations in the genes EIF2B1, EIF2B2, EIF2B3, EIF2B4, and EIF2B5 encoding eIF2B. We identified one novel nonsense mutation (EIF2B4, c.625C>T, p.Arg209X), one novel frameshift mutation (EIF2B5, c.453_454del, p.Tyr152fsX12), eight novel missense muations (EIF2B1, c.547G>T, p.Val183Phe; EIF2B2, c. 586C>T, p.Pro196Ser; EIF2B4, c.806T>G, p.Leu269Arg; EIF2B5, c.203T>C, p.Leu68Ser; EIF2B5, c.220G>A, p.Ala74Thr; EIF2B5, c.805C>G, p.Arg269Gly; EIF2B5, c.929G>T, p.Cys310Phe; EIF2B5, c.1003T>C, p.Cys335Arg), and eight previously described alterations.

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Year:  2005        PMID: 15776425     DOI: 10.1002/humu.9325

Source DB:  PubMed          Journal:  Hum Mutat        ISSN: 1059-7794            Impact factor:   4.878


  12 in total

Review 1.  Vanishing white matter disease: an Italian case with A638G mutation in exon 5 of EIF2B2 gene, an unusual early onset and a long course.

Authors:  Luisa Sambati; Raffaele Agati; Antonella Bacci; Silvia Bianchi; Sabina Capellari
Journal:  Neurol Sci       Date:  2012-06-23       Impact factor: 3.307

2.  Need for revision of the ACMG/AMP guidelines for interpretation of X-linked variants.

Authors:  Yoko Inoue; Osamu Machida; Yosuke Kita; Toshiyuki Yamamoto
Journal:  Intractable Rare Dis Res       Date:  2022-08

3.  Identification of a Novel Heterozygous Mutation in the EIF2B4 Gene Associated With Vanishing White Matter Disease.

Authors:  Yun Tian; Qiong Liu; Yafang Zhou; Xiao-Yu Chen; Yongcheng Pan; Hongwei Xu; Zhuanyi Yang
Journal:  Front Bioeng Biotechnol       Date:  2022-07-04

Review 4.  Genotypic and phenotypic characteristics of juvenile/adult onset vanishing white matter: a series of 14 Chinese patients.

Authors:  Yuting Ren; Xueying Yu; Bin Chen; Hefei Tang; Songtao Niu; Xingao Wang; Hua Pan; Zaiqiang Zhang
Journal:  Neurol Sci       Date:  2022-04-07       Impact factor: 3.830

5.  Sensitivity and specificity of decreased CSF asialotransferrin for eIF2B-related disorder.

Authors:  A Vanderver; Y Hathout; J Maletkovic; E S Gordon; M Mintz; M Timmons; E P Hoffman; L Horzinski; F Niel; A Fogli; O Boespflug-Tanguy; R Schiffmann
Journal:  Neurology       Date:  2008-06-03       Impact factor: 9.910

6.  Identification of residues that underpin interactions within the eukaryotic initiation factor (eIF2) 2B complex.

Authors:  Xuemin Wang; Noel C Wortham; Rui Liu; Christopher G Proud
Journal:  J Biol Chem       Date:  2012-01-11       Impact factor: 5.157

7.  Sugar phosphate activation of the stress sensor eIF2B.

Authors:  Qi Hao; Jin-Mi Heo; Boguslaw P Nocek; Kevin G Hicks; Vincent S Stoll; Clint Remarcik; Sean Hackett; Lauren LeBon; Rinku Jain; Dan Eaton; Jared Rutter; Yao Liang Wong; Carmela Sidrauski
Journal:  Nat Commun       Date:  2021-06-08       Impact factor: 14.919

8.  Fifteen novel EIF2B1-5 mutations identified in Chinese children with leukoencephalopathy with vanishing white matter and a long term follow-up.

Authors:  Haihua Zhang; Lifang Dai; Na Chen; Lili Zang; Xuerong Leng; Li Du; Jingmin Wang; Yuwu Jiang; Feng Zhang; Xiru Wu; Ye Wu
Journal:  PLoS One       Date:  2015-03-11       Impact factor: 3.240

9.  Biochemical effects of mutations in the gene encoding the alpha subunit of eukaryotic initiation factor (eIF) 2B associated with Vanishing White Matter disease.

Authors:  Noel C Wortham; Christopher G Proud
Journal:  BMC Med Genet       Date:  2015-08-19       Impact factor: 2.103

10.  Circadian transcriptome analysis in human fibroblasts from Hunter syndrome and impact of iduronate-2-sulfatase treatment.

Authors:  Gianluigi Mazzoccoli; Rosella Tomanin; Tommaso Mazza; Francesca D'Avanzo; Marika Salvalaio; Laura Rigon; Alessandra Zanetti; Valerio Pazienza; Massimo Francavilla; Francesco Giuliani; Manlio Vinciguerra; Maurizio Scarpa
Journal:  BMC Med Genomics       Date:  2013-10-02       Impact factor: 3.063

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