Literature DB >> 24218363

A new congenital disorder of glycosylation caused by a mutation in SSR4, the signal sequence receptor 4 protein of the TRAP complex.

Marie Estelle Losfeld1, Bobby G Ng, Martin Kircher, Kati J Buckingham, Emily H Turner, Alexey Eroshkin, Joshua D Smith, Jay Shendure, Deborah A Nickerson, Michael J Bamshad, Hudson H Freeze.   

Abstract

Nearly 50 congenital disorders of glycosylation (CDG) are known, but many patients biochemically diagnosed with CDG do not have mutations in known genes. Here, we describe a 16-year-old male who was born with microcephaly, developed intellectual disability, gastroesophageal reflux and a seizure disorder. We identified a de novo variant in the X-linked SSR4 gene which encodes a protein of the heterotetrameric translocon-associated protein (TRAP) complex. The c.316delT causes a p.F106Sfs*53 in SSR4 and also reduces expression of other TRAP complex proteins. The glycosylation marker Glyc-ER-GFP was used to confirm the underglycosylation in fibroblasts from the patient. Overexpression of the wild-type SSR4 allele partially restores glycosylation of the marker and of the other members of the TRAP complex. This is the first evidence that the TRAP complex, which binds to the oligosaccharyltransferase complex, is directly involved in N-glycosylation.

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Year:  2013        PMID: 24218363      PMCID: PMC3929095          DOI: 10.1093/hmg/ddt550

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


  16 in total

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2.  DDOST mutations identified by whole-exome sequencing are implicated in congenital disorders of glycosylation.

Authors:  Melanie A Jones; Bobby G Ng; Shruti Bhide; Ephrem Chin; Devin Rhodenizer; Ping He; Marie-Estelle Losfeld; Miao He; Kimiyo Raymond; Gerard Berry; Hudson H Freeze; Madhuri R Hegde
Journal:  Am J Hum Genet       Date:  2012-02-02       Impact factor: 11.025

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4.  A sensitive green fluorescent protein biomarker of N-glycosylation site occupancy.

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Journal:  FASEB J       Date:  2012-06-12       Impact factor: 5.191

Review 5.  Understanding human glycosylation disorders: biochemistry leads the charge.

Authors:  Hudson H Freeze
Journal:  J Biol Chem       Date:  2013-01-17       Impact factor: 5.157

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Authors:  Anita E Beck; Margaret J McMillin; Heidi I S Gildersleeve; Phillip R Kezele; Kathryn M Shively; John C Carey; Michael Regnier; Michael J Bamshad
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7.  DAD1 is required for the function and the structural integrity of the oligosaccharyltransferase complex.

Authors:  A Sanjay; J Fu; G Kreibich
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9.  Translocon-associated protein TRAP delta and a novel TRAP-like protein are coordinately expressed with pro-opiomelanocortin in Xenopus intermediate pituitary.

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Journal:  Biochem J       Date:  1995-11-15       Impact factor: 3.857

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  20 in total

Review 1.  Neurological aspects of human glycosylation disorders.

Authors:  Hudson H Freeze; Erik A Eklund; Bobby G Ng; Marc C Patterson
Journal:  Annu Rev Neurosci       Date:  2015-04-02       Impact factor: 12.449

Review 2.  Crosslinking and Reconstitution Approaches to Study Protein Transport.

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Journal:  Protein J       Date:  2019-06       Impact factor: 2.371

Review 3.  Components and Mechanisms of Import, Modification, Folding, and Assembly of Immunoglobulins in the Endoplasmic Reticulum.

Authors:  Richard Zimmermann
Journal:  J Clin Immunol       Date:  2016-02-29       Impact factor: 8.317

4.  Mutations in the translocon-associated protein complex subunit SSR3 cause a novel congenital disorder of glycosylation.

Authors:  Bobby G Ng; Charles M Lourenço; Marie-Estelle Losfeld; Kati J Buckingham; Martin Kircher; Deborah A Nickerson; Jay Shendure; Michael J Bamshad; Hudson H Freeze
Journal:  J Inherit Metab Dis       Date:  2019-04-16       Impact factor: 4.982

5.  Plasma cell deficiency in human subjects with heterozygous mutations in Sec61 translocon alpha 1 subunit (SEC61A1).

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Journal:  J Allergy Clin Immunol       Date:  2017-08-04       Impact factor: 10.793

Review 6.  What is new in CDG?

Authors:  Jaak Jaeken; Romain Péanne
Journal:  J Inherit Metab Dis       Date:  2017-05-08       Impact factor: 4.982

Review 7.  N-linked glycosylation and homeostasis of the endoplasmic reticulum.

Authors:  Natalia Cherepanova; Shiteshu Shrimal; Reid Gilmore
Journal:  Curr Opin Cell Biol       Date:  2016-04-14       Impact factor: 8.382

8.  Deficient endoplasmic reticulum translocon-associated protein complex limits the biosynthesis of proinsulin and insulin.

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9.  Expanding the Molecular and Clinical Phenotype of SSR4-CDG.

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Journal:  Hum Mutat       Date:  2015-08-27       Impact factor: 4.878

Review 10.  Perspectives on Glycosylation and Its Congenital Disorders.

Authors:  Bobby G Ng; Hudson H Freeze
Journal:  Trends Genet       Date:  2018-03-29       Impact factor: 11.639

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