Literature DB >> 33583022

Expanding the phenotype, genotype and biochemical knowledge of ALG3-CDG.

Hind Alsharhan1,2,3, Bobby G Ng4, Earnest James Paul Daniel2, Jennifer Friedman5, Eniko K Pivnick6, Amal Al-Hashem7,8, Eissa Ali Faqeih9, Pengfei Liu10,11, Nicole M Engelhardt1, Kierstin N Keller1, Jie Chen2, Pamela A Mazzeo12, Jill A Rosenfeld10,11, Michael J Bamshad13,14,15, Deborah A Nickerson14,15, Kimiyo M Raymond16, Hudson H Freeze4, Miao He2, Andrew C Edmondson1, Christina Lam13,17.   

Abstract

Congenital disorders of glycosylation (CDGs) are a continuously expanding group of monogenic disorders of glycoprotein and glycolipid biosynthesis that cause multisystem diseases. Individuals with ALG3-CDG frequently exhibit severe neurological involvement (epilepsy, microcephaly, and hypotonia), ocular anomalies, dysmorphic features, skeletal anomalies, and feeding difficulties. We present 10 unreported individuals diagnosed with ALG3-CDG based on molecular and biochemical testing with 11 novel variants in ALG3, bringing the total to 40 reported individuals. In addition to the typical multisystem disease seen in ALG3-CDG, we expand the symptomatology of ALG3-CDG to now include endocrine abnormalities, neural tube defects, mild aortic root dilatation, immunodeficiency, and renal anomalies. N-glycan analyses of these individuals showed combined deficiencies of hybrid glycans and glycan extension beyond Man5 GlcNAc2 consistent with their truncated lipid-linked precursor oligosaccharides. This spectrum of N-glycan changes is unique to ALG3-CDG. These expanded features of ALG3-CDG facilitate diagnosis and suggest that optimal management should include baseline endocrine, renal, cardiac, and immunological evaluation at the time of diagnosis and with ongoing monitoring.
© 2021 SSIEM.

Entities:  

Keywords:  N-glycans; congenital disorders of glycosylation; endocrine; immunodeficiency; neural tube defect

Mesh:

Substances:

Year:  2021        PMID: 33583022      PMCID: PMC8282734          DOI: 10.1002/jimd.12367

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.750


  34 in total

Review 1.  Integrating mass spectrometry of intact protein complexes into structural proteomics.

Authors:  Suk-Joon Hyung; Brandon T Ruotolo
Journal:  Proteomics       Date:  2012-05       Impact factor: 3.984

2.  Increased fucosylation and reduced branching of serum glycoprotein N-glycans in all known subtypes of congenital disorder of glycosylation I.

Authors:  Nico Callewaert; Els Schollen; Annelies Vanhecke; Jaak Jaeken; Gert Matthijs; Roland Contreras
Journal:  Glycobiology       Date:  2003-01-22       Impact factor: 4.313

3.  Carbohydrate deficient glycoprotein syndrome type IV: deficiency of dolichyl-P-Man:Man(5)GlcNAc(2)-PP-dolichyl mannosyltransferase.

Authors:  C Körner; R Knauer; U Stephani; T Marquardt; L Lehle; K von Figura
Journal:  EMBO J       Date:  1999-12-01       Impact factor: 11.598

4.  Increased Clinical Sensitivity and Specificity of Plasma Protein N-Glycan Profiling for Diagnosing Congenital Disorders of Glycosylation by Use of Flow Injection-Electrospray Ionization-Quadrupole Time-of-Flight Mass Spectrometry.

Authors:  Jie Chen; Xueli Li; Andrew Edmondson; Gail Ditewig Meyers; Kosuke Izumi; Amanda M Ackermann; Eva Morava; Can Ficicioglu; Michael J Bennett; Miao He
Journal:  Clin Chem       Date:  2019-02-15       Impact factor: 8.327

5.  Congenital disorders of glycosylation presenting as epileptic encephalopathy with migrating partial seizures in infancy.

Authors:  Carmen Barba; Francesca Darra; Raffaella Cusmai; Elena Procopio; Carlo Dionisi Vici; Liesbeth Keldermans; Sandrine Vuillaumier-Barrot; Dirk J Lefeber; Renzo Guerrini
Journal:  Dev Med Child Neurol       Date:  2016-05-13       Impact factor: 5.449

6.  CDG-Id caused by homozygosity for an ALG3 mutation due to segmental maternal isodisomy UPD3(q21.3-qter).

Authors:  E Schollen; S Grünewald; L Keldermans; B Albrecht; C Körner; G Matthijs
Journal:  Eur J Med Genet       Date:  2005-02-17       Impact factor: 2.708

7.  MRPL44 mutations cause a slowly progressive multisystem disease with childhood-onset hypertrophic cardiomyopathy.

Authors:  Felix Distelmaier; Tobias B Haack; Claudia B Catarino; Constanze Gallenmüller; Richard J Rodenburg; Tim M Strom; Fabian Baertling; Thomas Meitinger; Ertan Mayatepek; Holger Prokisch; Thomas Klopstock
Journal:  Neurogenetics       Date:  2015-03-24       Impact factor: 2.660

8.  Endocrinology of the carbohydrate-deficient glycoprotein syndrome type 1 from birth through adolescence.

Authors:  F de Zegher; J Jaeken
Journal:  Pediatr Res       Date:  1995-04       Impact factor: 3.756

9.  An activated 5' cryptic splice site in the human ALG3 gene generates a premature termination codon insensitive to nonsense-mediated mRNA decay in a new case of congenital disorder of glycosylation type Id (CDG-Id).

Authors:  Jonas Denecke; Christian Kranz; Dirk Kemming; Hans-Georg Koch; Thorsten Marquardt
Journal:  Hum Mutat       Date:  2004-05       Impact factor: 4.878

10.  Nijmegen paediatric CDG rating scale: a novel tool to assess disease progression.

Authors:  Samira Achouitar; Miski Mohamed; Thatjana Gardeitchik; Saskia B Wortmann; Jolanta Sykut-Cegielska; Regina Ensenauer; Hélène Ogier de Baulny; Katrin Õunap; Diego Martinelli; Maaike de Vries; Robert McFarland; Dorus Kouwenberg; Miranda Theodore; Frits Wijburg; Stephanie Grünewald; Jaak Jaeken; Ron A Wevers; Leo Nijtmans; Joanna Elson; Eva Morava
Journal:  J Inherit Metab Dis       Date:  2011-05-04       Impact factor: 4.982

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  3 in total

1.  Expression of ALG3 in Hepatocellular Carcinoma and Its Clinical Implication.

Authors:  Zhen Zhao; Zehao Zheng; Jianfeng Huang; Jianxi Wang; Tianyi Peng; Ye Lin; Zhixiang Jian
Journal:  Front Mol Biosci       Date:  2022-06-15

Review 2.  ALG3-CDG: a patient with novel variants and review of the genetic and ophthalmic findings.

Authors:  Martina Farolfi; Anna Cechova; Nina Ondruskova; Jana Zidkova; Bohdan Kousal; Hana Hansikova; Tomas Honzik; Petra Liskova
Journal:  BMC Ophthalmol       Date:  2021-06-05       Impact factor: 2.209

3.  Synergistic use of glycomics and single-molecule molecular inversion probes for identification of congenital disorders of glycosylation type-1.

Authors:  Nurulamin Abu Bakar; Angel Ashikov; Jaime Moritz Brum; Roel Smeets; Marjan Kersten; Karin Huijben; Wee Teik Keng; Carlos Eduardo Speck-Martins; Daniel Rocha de Carvalho; Isabela Maria Pinto Oliveira de Rizzo; Walquiria Domingues de Mello; Rebecca Heiner-Fokkema; Kathleen Gorman; Stephanie Grunewald; Helen Michelakakis; Marina Moraitou; Diego Martinelli; Monique van Scherpenzeel; Mirian Janssen; Lonneke de Boer; Lambertus P van den Heuvel; Christian Thiel; Dirk J Lefeber
Journal:  J Inherit Metab Dis       Date:  2022-03-28       Impact factor: 4.750

  3 in total

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