Literature DB >> 21498477

Identification of a novel candidate gene for non-syndromic autosomal recessive intellectual disability: the WASH complex member SWIP.

Fabienne Ropers1, Emmanuel Derivery, Hao Hu, Masoud Garshasbi, Mohsen Karbasiyan, Martin Herold, Gudrun Nürnberg, Reinhard Ullmann, Alexis Gautreau, Karl Sperling, Raymonda Varon, Anna Rajab.   

Abstract

High-throughput sequencing has greatly facilitated the elucidation of genetic disorders, but compared with X-linked and autosomal dominant diseases, the search for genetic defects underlying autosomal recessive diseases still lags behind. In a large consanguineous family with autosomal recessive intellectual disability (ARID), we have combined homozygosity mapping, targeted exon enrichment and high-throughput sequencing to identify the underlying gene defect. After appropriate single-nucleotide polymorphism filtering, only two molecular changes remained, including a non-synonymous sequence change in the SWIP [Strumpellin and WASH (Wiskott-Aldrich syndrome protein and scar homolog)-interacting protein] gene, a member of the recently discovered WASH complex, which is involved in actin polymerization and multiple endosomal transport processes. Based on high pathogenicity and evolutionary conservation scores as well as functional considerations, this gene defect was considered as causative for ID in this family. In line with this assumption, we could show that this mutation leads to significantly reduced SWIP levels and to destabilization of the entire WASH complex. Thus, our findings suggest that SWIP is a novel gene for ARID.

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Year:  2011        PMID: 21498477     DOI: 10.1093/hmg/ddr158

Source DB:  PubMed          Journal:  Hum Mol Genet        ISSN: 0964-6906            Impact factor:   6.150


  35 in total

Review 1.  Actin Out: Regulation of the Synaptic Cytoskeleton.

Authors:  Erin F Spence; Scott H Soderling
Journal:  J Biol Chem       Date:  2015-10-09       Impact factor: 5.157

Review 2.  The retromer complex - endosomal protein recycling and beyond.

Authors:  Matthew N J Seaman
Journal:  J Cell Sci       Date:  2012-11-12       Impact factor: 5.285

Review 3.  Function and regulation of the endosomal fusion and fission machineries.

Authors:  Alexis Gautreau; Ksenia Oguievetskaia; Christian Ungermann
Journal:  Cold Spring Harb Perspect Biol       Date:  2014-03-01       Impact factor: 10.005

4.  RME-8 coordinates the activity of the WASH complex with the function of the retromer SNX dimer to control endosomal tubulation.

Authors:  Caroline L Freeman; Geoffrey Hesketh; Matthew N J Seaman
Journal:  J Cell Sci       Date:  2014-03-18       Impact factor: 5.285

5.  Expanding the clinical and mutational spectrum of Kaufman oculocerebrofacial syndrome with biallelic UBE3B mutations.

Authors:  Lina Basel-Vanagaite; Rüstem Yilmaz; Sha Tang; Miriam S Reuter; Nils Rahner; Dorothy K Grange; Megan Mortenson; Patrick Koty; Heather Feenstra; Kelly D Farwell Gonzalez; Heinrich Sticht; Nathalie Boddaert; Julie Désir; Kwame Anyane-Yeboa; Christiane Zweier; André Reis; Christian Kubisch; Tamison Jewett; Wenqi Zeng; Guntram Borck
Journal:  Hum Genet       Date:  2014-03-11       Impact factor: 4.132

6.  Drosophila Wash and the Wash regulatory complex function in nuclear envelope budding.

Authors:  Jeffrey M Verboon; Mitsutoshi Nakamura; Kerri A Davidson; Jacob R Decker; Vivek Nandakumar; Susan M Parkhurst
Journal:  J Cell Sci       Date:  2020-07-08       Impact factor: 5.285

7.  The hepatic WASH complex is required for efficient plasma LDL and HDL cholesterol clearance.

Authors:  Melinde Wijers; Paolo Zanoni; Nalan Liv; Dyonne Y Vos; Michelle Y Jäckstein; Marieke Smit; Sanne Wilbrink; Justina C Wolters; Ydwine T van der Veen; Nicolette Huijkman; Daphne Dekker; Niels Kloosterhuis; Theo H van Dijk; Daniel D Billadeau; Folkert Kuipers; Judith Klumperman; Arnold von Eckardstein; Jan Albert Kuivenhoven; Bart van de Sluis
Journal:  JCI Insight       Date:  2019-06-06

8.  Nuclear FAM21 participates in NF-κB-dependent gene regulation in pancreatic cancer cells.

Authors:  Zhi-Hui Deng; Timothy S Gomez; Douglas G Osborne; Christine A Phillips-Krawczak; Jin-San Zhang; Daniel D Billadeau
Journal:  J Cell Sci       Date:  2014-11-27       Impact factor: 5.285

9.  Wash exhibits context-dependent phenotypes and, along with the WASH regulatory complex, regulates Drosophila oogenesis.

Authors:  Jeffrey M Verboon; Jacob R Decker; Mitsutoshi Nakamura; Susan M Parkhurst
Journal:  J Cell Sci       Date:  2018-04-13       Impact factor: 5.285

Review 10.  Emerging roles of the MAGE protein family in stress response pathways.

Authors:  Rebecca R Florke Gee; Helen Chen; Anna K Lee; Christina A Daly; Benjamin A Wilander; Klementina Fon Tacer; Patrick Ryan Potts
Journal:  J Biol Chem       Date:  2020-09-13       Impact factor: 5.157

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