Literature DB >> 31481313

Complex phenotypes in ALG12-congenital disorder of glycosylation (ALG12-CDG): Case series and review of the literature.

Shawn Tahata1, Lauren Gunderson1, Brendan Lanpher1, Eva Morava2.   

Abstract

ALG12-congenital disorder of glycosylation (ALG12-CDG) is a rare disorder caused by a deficiency of dolichol-P-mannose:Man7GlcNAc2-PP-dolichyl-α-6-mannosyltransferase which presents with intellectual disability, hypotonia, dysmorphic features, low IgG levels with recurrent infections, male genital hypoplasia, and coagulation abnormalities. We report a unique family with three affected individuals, including two older brothers with only cognitive and coagulation defects and a younger brother who died from a severe multisystem disease at age 18 months. The two living brothers are the oldest and mildest cases of ALG12-CDG described thus far. Whole exome sequencing of the older brothers revealed a previously described c.1001delA (p.N334TfsX15) pathogenic variant and a c.671C > T (p.T224 M) variant of uncertain significance in ALG12. Our cases broaden the recognized genetic and phenotypic spectrum of this disorder and suggest a role for other genetic and environmental factors in modulating disease phenotype.
Copyright © 2019 Elsevier Inc. All rights reserved.

Entities:  

Keywords:  ALG12; ALG12-CDG; CDG; Congenital disorder of glycosylation

Mesh:

Substances:

Year:  2019        PMID: 31481313     DOI: 10.1016/j.ymgme.2019.08.007

Source DB:  PubMed          Journal:  Mol Genet Metab        ISSN: 1096-7192            Impact factor:   4.797


  6 in total

1.  A Novel Homozygous ALG12 Mutation in a Patient with CDG Type Ig: New Report of a Case with a Mild Phenotype.

Authors:  Antonio Gennaro Nicotera; Giulia Spoto; Francesco Calì; Giusi Romeo; Antonino Musumeci; Mirella Vinci; Agata Fiumara; Rita Barone; Gabriella Di Rosa; Sebastiano Antonino Musumeci
Journal:  Mol Syndromol       Date:  2021-07-20

2.  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

3.  A novel homozygous mutation in the human ALG12 gene results in an aberrant profile of oligomannose N-glycans in patient's serum.

Authors:  Jana Ziburová; Marek Nemčovič; Sergej Šesták; Jana Bellová; Zuzana Pakanová; Barbara Siváková; Anna Šalingová; Claudia Šebová; Mária Ostrožlíková; Dimitra-Evanthia Lekka; Jana Brucknerová; Ingrid Brucknerová; Martina Skokňová; Alexandra Mc Cullough; Gabriela Hrčková; Anna Hlavatá; Vladimír Bzdúch; Ján Mucha; Peter Baráth
Journal:  Am J Med Genet A       Date:  2021-09-01       Impact factor: 2.578

Review 4.  Genotype-phenotype correlation in Phelan-McDermid syndrome: A comprehensive review of chromosome 22q13 deleted genes.

Authors:  Arianna Ricciardello; Pasquale Tomaiuolo; Antonio M Persico
Journal:  Am J Med Genet A       Date:  2021-05-05       Impact factor: 2.802

5.  Liver Involvement in Congenital Disorders of Glycosylation: A Systematic Review.

Authors:  Rossella Colantuono; Elisa D'Acunto; Daniela Melis; Pietro Vajro; Hudson H Freeze; Claudia Mandato
Journal:  J Pediatr Gastroenterol Nutr       Date:  2021-10-01       Impact factor: 3.288

6.  ALG12-CDG: An unusual patient without intellectual disability and facial dysmorphism, and with a novel variant.

Authors:  María Eugenia de la Morena-Barrio; María Sabater; Belén de la Morena-Barrio; Renee L Ruhaak; Antonia Miñano; José Padilla; Mara Toderici; Vanessa Roldán; Juan R Gimeno; Vicente Vicente; Javier Corral
Journal:  Mol Genet Genomic Med       Date:  2020-06-12       Impact factor: 2.183

  6 in total

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