Literature DB >> 33393008

Clinical and molecular findings in a Turkish family with an ultra-rare condition, ELP2-related neurodevelopmental disorder.

Mustafa Dogan1, Kerem Teralı2, Recep Eroz3, Huseyin Demirci4, Kenan Kocabay5.   

Abstract

Elongator is a multi-subunit protein complex bearing six different protein subunits, Elp1 to -6, that are highly conserved among eukaryotes. Elp2 is the second major subunit of Elongator and, together with Elp1 and Elp3, form the catalytic core of this essential complex. Pathogenic variants that affect the structure and function of the Elongator complex may cause neurodevelopmental disorders. Here, we report on a new family with three children affected with a severe form of intellectual disability along with spastic tetraparesis, choreoathetosis, and self injury. Molecular genetic analyses reveal a homozygous missense variant in the ELP2 gene (NM_018255.4 (ELP2): c.1385G > A (p.Arg462Gln)), while in silico studies suggest a loss of electrostatic interactions that may contribute to the overall stability of the encoded protein. We also include a comparison of the patients with ELP2-related neurodevelopmental disorder to those previously reported in the literature. Apart from being affected with intellectual disability, we have extremely limited clinical knowledge about patients harboring ELP2 variants. Besides providing support to the causal role of p.Arg462Gln in ELP2-related neurodevelopmental disorder, we add self-injurious behavior to the clinical phenotypic repertoire of the disease.

Entities:  

Keywords:  Choreoathetosis; ELP2; Elongator complex; Intellectual disability; Self injury; Spasticity

Mesh:

Substances:

Year:  2021        PMID: 33393008     DOI: 10.1007/s11033-020-06097-9

Source DB:  PubMed          Journal:  Mol Biol Rep        ISSN: 0301-4851            Impact factor:   2.316


  14 in total

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Journal:  Nature       Date:  2011-09-21       Impact factor: 49.962

2.  A Scalable Genome-Editing-Based Approach for Mapping Multiprotein Complexes in Human Cells.

Authors:  Mathieu Dalvai; Jeremy Loehr; Karine Jacquet; Caroline C Huard; Céline Roques; Pauline Herst; Jacques Côté; Yannick Doyon
Journal:  Cell Rep       Date:  2015-10-08       Impact factor: 9.423

3.  Elongator controls the migration and differentiation of cortical neurons through acetylation of alpha-tubulin.

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Journal:  Cell       Date:  2009-01-29       Impact factor: 41.582

4.  HomSI: a homozygous stretch identifier from next-generation sequencing data.

Authors:  Zeliha Görmez; Burcu Bakir-Gungor; Mahmut Samil Sagiroglu
Journal:  Bioinformatics       Date:  2013-12-03       Impact factor: 6.937

5.  The Elp2 subunit is essential for elongator complex assembly and functional regulation.

Authors:  Chunming Dong; Zhijie Lin; Wentao Diao; Dan Li; Xinlei Chu; Zheng Wang; Hao Zhou; Zhiping Xie; Yuequan Shen; Jiafu Long
Journal:  Structure       Date:  2015-05-07       Impact factor: 5.006

6.  FMFilter: A fast model based variant filtering tool.

Authors:  Mete Akgün; Ö Faruk Gerdan; Zeliha Görmez; Hüseyin Demirci
Journal:  J Biomed Inform       Date:  2016-02-27       Impact factor: 6.317

7.  Transcription impairment and cell migration defects in elongator-depleted cells: implication for familial dysautonomia.

Authors:  Pierre Close; Nicola Hawkes; Isabelle Cornez; Catherine Creppe; Charles A Lambert; Bernard Rogister; Ulrich Siebenlist; Marie-Paule Merville; Susan A Slaugenhaupt; Vincent Bours; Jesper Q Svejstrup; Alain Chariot
Journal:  Mol Cell       Date:  2006-05-19       Impact factor: 17.970

Review 8.  Structural insights into the function of Elongator.

Authors:  Udit Dalwadi; Calvin K Yip
Journal:  Cell Mol Life Sci       Date:  2018-01-13       Impact factor: 9.261

9.  Microdeletions of ELP4 Are Associated with Language Impairment, Autism Spectrum Disorder, and Mental Retardation.

Authors:  Laura Addis; Joo Wook Ahn; Richard Dobson; Abhishek Dixit; Caroline M Ogilvie; Dalila Pinto; Andrea K Vaags; Hilary Coon; Pauline Chaste; Scott Wilson; Jeremy R Parr; Joris Andrieux; Bruno Lenne; Zeynep Tumer; Vincenzo Leuzzi; Kristina Aubell; Hannele Koillinen; Sarah Curran; Christian R Marshall; Stephen W Scherer; Lisa J Strug; David A Collier; Deb K Pal
Journal:  Hum Mutat       Date:  2015-06-30       Impact factor: 4.878

10.  Architecture of the yeast Elongator complex.

Authors:  Maria I Dauden; Jan Kosinski; Olga Kolaj-Robin; Ambroise Desfosses; Alessandro Ori; Celine Faux; Niklas A Hoffmann; Osita F Onuma; Karin D Breunig; Martin Beck; Carsten Sachse; Bertrand Séraphin; Sebastian Glatt; Christoph W Müller
Journal:  EMBO Rep       Date:  2016-12-14       Impact factor: 8.807

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