Literature DB >> 28777499

ALG13-CDG in a male with seizures, normal cognitive development, and normal transferrin isoelectric focusing.

Therese E Gadomski1,2, Melody Bolton1, Majid Alfadhel3, Chris Dvorak1, Olalekan A Ogunsakin1, Stephen L Nelson2, Eva Morava1,2.   

Abstract

ALG13-CDG has been recently discovered as a disorder of severe developmental, intellectual and speech disability, microcephaly, visual abnormalities, seizures, hepatomegaly, coagulation abnormalities, and abnormal serumtransferrin isoelectric focusing in serum. A male with seizures, delayed motor, and speech development, but normal cognition carried a hemizygous, predicted pathogenic ALG13 variant (p.E463G). N-glycosylation studies in plasma were normal. ICAM-1 expression was decreased in patient fibroblasts, supporting the variant's pathogenicity. Adding D-galactose to the patient's fibroblast culture increased ICAM-1 expression in vitro, offering a potential treatment option in ALG13-CDG. The present report is a new example for an N-glycosylation disorder, that may present with normal transferrin isoform analysis, and also demonstrates, that CDG type I patients can have normal cognitive development.
© 2017 Wiley Periodicals, Inc.

Entities:  

Keywords:  ICAM-1; galactose; glycosylation; normal TIEF; seizures

Mesh:

Substances:

Year:  2017        PMID: 28777499     DOI: 10.1002/ajmg.a.38377

Source DB:  PubMed          Journal:  Am J Med Genet A        ISSN: 1552-4825            Impact factor:   2.802


  9 in total

1.  Neurological Consequences of Congenital Disorders of Glycosylation.

Authors:  Justyna Paprocka
Journal:  Adv Neurobiol       Date:  2023

Review 2.  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

3.  ALG13 X-linked intellectual disability: New variants, glycosylation analysis, and expanded phenotypes.

Authors:  Hind Alsharhan; Miao He; Andrew C Edmondson; Earnest J P Daniel; Jie Chen; Tyhiesia Donald; Somayeh Bakhtiari; David J Amor; Elizabeth A Jones; Grace Vassallo; Marie Vincent; Benjamin Cogné; Wallid Deb; Arend H Werners; Sheng C Jin; Kaya Bilguvar; John Christodoulou; Richard I Webster; Katherine R Yearwood; Bobby G Ng; Hudson H Freeze; Michael C Kruer; Dong Li; Kimiyo M Raymond; Elizabeth J Bhoj; Andrew K Sobering
Journal:  J Inherit Metab Dis       Date:  2021-03-26       Impact factor: 4.750

Review 4.  CDG Therapies: From Bench to Bedside.

Authors:  Sandra Brasil; Carlota Pascoal; Rita Francisco; Dorinda Marques-da-Silva; Giuseppina Andreotti; Paula A Videira; Eva Morava; Jaak Jaeken; Vanessa Dos Reis Ferreira
Journal:  Int J Mol Sci       Date:  2018-04-27       Impact factor: 5.923

5.  The phenotypic spectrum of X-linked, infantile onset ALG13-related developmental and epileptic encephalopathy.

Authors:  Alexandre N Datta; Nadia Bahi-Buisson; Thierry Bienvenu; Sarah E Buerki; Fiona Gardiner; J Helen Cross; Bénédicte Heron; Anna Kaminska; Christian M Korff; Anne Lepine; Gaetan Lesca; Amy McTague; Heather C Mefford; Cyrill Mignot; Matthieu Milh; Amélie Piton; Ronit M Pressler; Susanne Ruf; Lynette G Sadleir; Anne de Saint Martin; Koen Van Gassen; Nienke E Verbeek; Dorothée Ville; Nathalie Villeneuve; Pia Zacher; Ingrid E Scheffer; Johannes R Lemke
Journal:  Epilepsia       Date:  2021-01-07       Impact factor: 5.864

Review 6.  Congenital Disorders of Glycosylation from a Neurological Perspective.

Authors:  Justyna Paprocka; Aleksandra Jezela-Stanek; Anna Tylki-Szymańska; Stephanie Grunewald
Journal:  Brain Sci       Date:  2021-01-11

7.  Case Report: Identification of Two Variants of ALG13 in Families With or Without Seizure and Binocular Strabismus: Phenotypic Spectrum Analysis.

Authors:  Tao Cai; Jieting Huang; Xiuwei Ma; Siqi Hu; Lina Zhu; Jinwen Zhu; Zhichun Feng
Journal:  Front Genet       Date:  2022-07-11       Impact factor: 4.772

8.  An in vitro assay for enzymatic studies on human ALG13/14 heterodimeric UDP-N-acetylglucosamine transferase.

Authors:  Chun-Di Wang; Si Xu; Shuai Chen; Zheng-Hui Chen; Neta Dean; Ning Wang; Xiao-Dong Gao
Journal:  Front Cell Dev Biol       Date:  2022-09-19

9.  Structural Analysis of the Effect of Asn107Ser Mutation on Alg13 Activity and Alg13-Alg14 Complex Formation and Expanding the Phenotypic Variability of ALG13-CDG.

Authors:  Karolina Mitusińska; Artur Góra; Anna Bogdańska; Agnieszka Rożdżyńska-Świątkowska; Anna Tylki-Szymańska; Aleksandra Jezela-Stanek
Journal:  Biomolecules       Date:  2022-03-04
  9 in total

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