Literature DB >> 23430903

Deficiency of Subunit 6 of the Conserved Oligomeric Golgi Complex (COG6-CDG): Second Patient, Different Phenotype.

S Huybrechts1, C De Laet, P Bontems, S Rooze, H Souayah, Y Sznajer, L Sturiale, D Garozzo, G Matthijs, A Ferster, J Jaeken, P Goyens.   

Abstract

We describe a 27-month-old girl with COG6 deficiency. She is the first child of healthy consanguineous Moroccan parents. She presented at birth with dysmorphic features including microcephaly, post-axial polydactyly, broad palpebral fissures, retrognathia, and anal anteposition. The clinical phenotype was further characterised by multiorgan involvement including mild psychomotor retardation, and microcephaly, chronic inflammatory bowel disease, micronodular liver cirrhosis, associated with life-threatening and recurrent infections due to combined T- and B-cell dysfunction and neutrophil dysfunction.Mutation analysis showed the patient to be homozygous for the c.G1646T mutation in the COG6 gene. She is the second reported patient with a deficiency of subunit 6 of the COG complex. Although both patients are homozygous for the same mutation, they present a markedly different clinical picture. Indeed immunodeficiency as well as inflammatory bowel disease has not been described previously in patients with any COG-CDG.

Entities:  

Year:  2011        PMID: 23430903      PMCID: PMC3509899          DOI: 10.1007/8904_2011_79

Source DB:  PubMed          Journal:  JIMD Rep        ISSN: 2192-8304


  26 in total

1.  Clinical and biochemical presentation of siblings with COG-7 deficiency, a lethal multiple O- and N-glycosylation disorder.

Authors:  L J M Spaapen; J A Bakker; S B van der Meer; H J Sijstermans; R A Steet; R A Wevers; J Jaeken
Journal:  J Inherit Metab Dis       Date:  2005       Impact factor: 4.982

2.  A new inborn error of glycosylation due to a Cog8 deficiency reveals a critical role for the Cog1-Cog8 interaction in COG complex formation.

Authors:  François Foulquier; Daniel Ungar; Ellen Reynders; Renate Zeevaert; Philippa Mills; Maria Teresa García-Silva; Paz Briones; Bryan Winchester; Willy Morelle; Monty Krieger; Willem Annaert; Gert Matthijs
Journal:  Hum Mol Genet       Date:  2007-01-12       Impact factor: 6.150

3.  Recurrent infections and immunological dysfunction in congenital disorder of glycosylation Ia (CDG Ia).

Authors:  C Blank; L A Smith; D A Hammer; M Fehrenbach; H M Delisser; E Perez; K E Sullivan
Journal:  J Inherit Metab Dis       Date:  2006-07-06       Impact factor: 4.982

4.  A common mutation in the COG7 gene with a consistent phenotype including microcephaly, adducted thumbs, growth retardation, VSD and episodes of hyperthermia.

Authors:  Eva Morava; Renate Zeevaert; Eckhard Korsch; Karin Huijben; Suzan Wopereis; Gert Matthijs; Kathelijn Keymolen; Dirk J Lefeber; Linda De Meirleir; Ron A Wevers
Journal:  Eur J Hum Genet       Date:  2007-03-14       Impact factor: 4.246

5.  COG8 deficiency causes new congenital disorder of glycosylation type IIh.

Authors:  Christian Kranz; Bobby G Ng; Liangwu Sun; Vandana Sharma; Erik A Eklund; Yoshiaki Miura; Daniel Ungar; Vladimir Lupashin; R Dennis Winkel; John F Cipollo; Catherine E Costello; Eva Loh; Wanjin Hong; Hudson H Freeze
Journal:  Hum Mol Genet       Date:  2007-03-01       Impact factor: 6.150

6.  Conserved oligomeric Golgi complex subunit 1 deficiency reveals a previously uncharacterized congenital disorder of glycosylation type II.

Authors:  François Foulquier; Eliza Vasile; Els Schollen; Nico Callewaert; Tim Raemaekers; Dulce Quelhas; Jaak Jaeken; Philippa Mills; Bryan Winchester; Monty Krieger; Wim Annaert; Gert Matthijs
Journal:  Proc Natl Acad Sci U S A       Date:  2006-02-28       Impact factor: 11.205

7.  Mutation of the COG complex subunit gene COG7 causes a lethal congenital disorder.

Authors:  Xiaohua Wu; Richard A Steet; Ognian Bohorov; Jaap Bakker; John Newell; Monty Krieger; Leo Spaapen; Stuart Kornfeld; Hudson H Freeze
Journal:  Nat Med       Date:  2004-04-25       Impact factor: 53.440

8.  Purified cytochrome b from human granulocyte plasma membrane is comprised of two polypeptides with relative molecular weights of 91,000 and 22,000.

Authors:  C A Parkos; R A Allen; C G Cochrane; A J Jesaitis
Journal:  J Clin Invest       Date:  1987-09       Impact factor: 14.808

9.  Molecular and clinical characterization of a Moroccan Cog7 deficient patient.

Authors:  Bobby G Ng; Christian Kranz; E E O Hagebeuk; M Duran; N G G M Abeling; B Wuyts; Daniel Ungar; Vladimir Lupashin; C M Hartdorff; B T Poll-The; Hudson H Freeze
Journal:  Mol Genet Metab       Date:  2007-03-28       Impact factor: 4.797

Review 10.  Congenital disorders of glycosylation: a rapidly expanding disease family.

Authors:  Jaak Jaeken; Gert Matthijs
Journal:  Annu Rev Genomics Hum Genet       Date:  2007       Impact factor: 8.929

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  14 in total

1.  Secondary Hemophagocytic Syndrome Associated with COG6 Gene Defect: Report and Review.

Authors:  Nouf Althonaian; Abdulrahman Alsultan; Eva Morava; Majid Alfadhel
Journal:  JIMD Rep       Date:  2018-02-15

Review 2.  Conserved Oligomeric Golgi and Neuronal Vesicular Trafficking.

Authors:  Leslie K Climer; Rachel D Hendrix; Vladimir V Lupashin
Journal:  Handb Exp Pharmacol       Date:  2018

Review 3.  Genetics of inflammatory bowel disease from multifactorial to monogenic forms.

Authors:  Anna Monica Bianco; Martina Girardelli; Alberto Tommasini
Journal:  World J Gastroenterol       Date:  2015-11-21       Impact factor: 5.742

Review 4.  Immunological aspects of congenital disorders of glycosylation (CDG): a review.

Authors:  Maria Monticelli; Tiago Ferro; Jaak Jaeken; Vanessa Dos Reis Ferreira; Paula A Videira
Journal:  J Inherit Metab Dis       Date:  2016-07-08       Impact factor: 4.750

Review 5.  Bridging the Gap between Glycosylation and Vesicle Traffic.

Authors:  Peter Fisher; Daniel Ungar
Journal:  Front Cell Dev Biol       Date:  2016-03-08

6.  The interactome of the copper transporter ATP7A belongs to a network of neurodevelopmental and neurodegeneration factors.

Authors:  Heather S Comstra; Jacob McArthy; Samantha Rudin-Rush; Cortnie Hartwig; Avanti Gokhale; Stephanie A Zlatic; Jessica B Blackburn; Erica Werner; Michael Petris; Priya D'Souza; Parinya Panuwet; Dana Boyd Barr; Vladimir Lupashin; Alysia Vrailas-Mortimer; Victor Faundez
Journal:  Elife       Date:  2017-03-29       Impact factor: 8.140

Review 7.  Clinical glycomics for the diagnosis of congenital disorders of glycosylation.

Authors:  Nurulamin Abu Bakar; Dirk J Lefeber; Monique van Scherpenzeel
Journal:  J Inherit Metab Dis       Date:  2018-03-01       Impact factor: 4.982

Review 8.  Sugary Logistics Gone Wrong: Membrane Trafficking and Congenital Disorders of Glycosylation.

Authors:  Peter T A Linders; Ella Peters; Martin Ter Beest; Dirk J Lefeber; Geert van den Bogaart
Journal:  Int J Mol Sci       Date:  2020-06-30       Impact factor: 5.923

Review 9.  Defects in the COG complex and COG-related trafficking regulators affect neuronal Golgi function.

Authors:  Leslie K Climer; Maxim Dobretsov; Vladimir Lupashin
Journal:  Front Neurosci       Date:  2015-10-27       Impact factor: 4.677

Review 10.  Maintaining order: COG complex controls Golgi trafficking, processing, and sorting.

Authors:  Jessica B Blackburn; Zinia D'Souza; Vladimir V Lupashin
Journal:  FEBS Lett       Date:  2019-08-16       Impact factor: 4.124

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