Literature DB >> 35716054

ALG8-CDG: Molecular and phenotypic expansion suggests clinical management guidelines.

Daniah Albokhari1,2, Bobby G Ng3, Alis Guberinic4, Earnest James Paul Daniel5, Nicole M Engelhardt1, Rita Barone6, Agata Fiumara7, Livia Garavelli8, Gabriele Trimarchi8, Lynne Wolfe9, Kimiyo M Raymond10, Eva Morava4, Miao He5, Hudson H Freeze3, Christina Lam11,12, Andrew C Edmondson1.   

Abstract

Congenital disorders of glycosylation are a continuously expanding group of monogenic disorders of glycoprotein and glycolipid glycan biosynthesis. These disorders mostly manifest with multisystem involvement. Individuals with ALG8-CDG commonly present with hypotonia, protein-losing enteropathy, and hepatic involvement. Here, we describe seven unreported individuals diagnosed with ALG8-CDG based on biochemical and molecular testing and we identify nine novel variants in ALG8, bringing the total to 26 individuals with ALG8-CDG in the medical literature. In addition to the typical multisystem involvement documented in ALG8-CDG, our cohort includes the two oldest patients reported and further expands the phenotype of ALG8-CDG to include stable intellectual disability, autism spectrum disorder and other neuropsychiatric symptoms. We further expand the clinical features in a variety of organ systems including ocular, musculoskeletal, dermatologic, endocrine, and cardiac abnormalities and suggest a comprehensive evaluation and monitoring strategy to improve clinical management.
© 2022 SSIEM.

Entities:  

Keywords:  N-glycans; congenital disorders of glycosylation; lipid-linked oligosaccharides

Mesh:

Substances:

Year:  2022        PMID: 35716054      PMCID: PMC9474684          DOI: 10.1002/jimd.12527

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


  37 in total

1.  Severe ALG8-CDG (CDG-Ih) associated with homozygosity for two novel missense mutations detected by exome sequencing of candidate genes.

Authors:  Hanne Sorte; Lars Mørkrid; Olaug Rødningen; Mari Ann Kulseth; Asbjørg Stray-Pedersen; Gert Matthijs; Valerie Race; Gunnar Houge; Torunn Fiskerstrand; Bjørn Bjurulf; Robert Lyle; Trine Prescott
Journal:  Eur J Med Genet       Date:  2012-01-16       Impact factor: 2.708

2.  Lymphatic edema in congenital disorders of glycosylation.

Authors:  Ruud Hj Verstegen; Miranda Theodore; Hans van de Klerk; Eva Morava
Journal:  JIMD Rep       Date:  2011-10-20

3.  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

4.  CDG nomenclature: time for a change!

Authors:  Jaak Jaeken; Thierry Hennet; Gert Matthijs; Hudson H Freeze
Journal:  Biochim Biophys Acta       Date:  2009-09

5.  Novel ALG8 mutations expand the clinical spectrum of congenital disorder of glycosylation type Ih.

Authors:  Torsten Stölting; Heymut Omran; Anne Erlekotte; Jonas Denecke; Janine Reunert; Thorsten Marquardt
Journal:  Mol Genet Metab       Date:  2009-06-24       Impact factor: 4.797

6.  Wide clinical spectrum in ALG8-CDG: clues from molecular findings suggest an explanation for a milder phenotype in the first-described patient.

Authors:  Sandrine Vuillaumier-Barrot; Manuel Schiff; Francesca Mattioli; Elise Schaefer; Audrey Dupont; Julia Dancourt; Thierry Dupré; Alain Couvineau; Hélène Ogier de Baulny; Pascale de Lonlay; Nathalie Seta; Stuart Moore; Isabelle Chantret
Journal:  Pediatr Res       Date:  2018-11-12       Impact factor: 3.756

7.  Anesthetic management of a child with phosphomannomutase-2 congenital disorder of glycosylation (PMM2-CDG).

Authors:  Wataru Sakai; Yusuke Yoshikawa; Yasuyuki Tokinaga; Michiaki Yamakage
Journal:  JA Clin Rep       Date:  2017-02-10

8.  Liver manifestations in a cohort of 39 patients with congenital disorders of glycosylation: pin-pointing the characteristics of liver injury and proposing recommendations for follow-up.

Authors:  Rodrigo Tzovenos Starosta; Suzanne Boyer; Shawn Tahata; Kimiyo Raymond; Hee Eun Lee; Lynne A Wolfe; Christina Lam; Andrew C Edmondson; Ida Vanessa Doederlein Schwartz; Eva Morava
Journal:  Orphanet J Rare Dis       Date:  2021-01-07       Impact factor: 4.123

9.  VarSome: the human genomic variant search engine.

Authors:  Christos Kopanos; Vasilis Tsiolkas; Alexandros Kouris; Charles E Chapple; Monica Albarca Aguilera; Richard Meyer; Andreas Massouras
Journal:  Bioinformatics       Date:  2019-06-01       Impact factor: 6.937

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