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N-Glycoprofiling of SLC35A2-CDG: Patient with a Novel Hemizygous Variant

Congenital disorders of glycosylation (CDG) are a group of rare inherited metabolic disorders caused by a defect in the process of protein glycosylation. In this work, we present a comprehensive glycoprofile analysis of a male patient with a novel missense variant in the SLC35A2 gene, coding a galac...

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Autores principales: Kodríková, Rebeka, Pakanová, Zuzana, Krchňák, Maroš, Šedivá, Mária, Šesták, Sergej, Květoň, Filip, Beke, Gábor, Šalingová, Anna, Skalická, Katarína, Brennerová, Katarína, Jančová, Emília, Baráth, Peter, Mucha, Ján, Nemčovič, Marek
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9952902/
https://www.ncbi.nlm.nih.gov/pubmed/36831116
http://dx.doi.org/10.3390/biomedicines11020580
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author Kodríková, Rebeka
Pakanová, Zuzana
Krchňák, Maroš
Šedivá, Mária
Šesták, Sergej
Květoň, Filip
Beke, Gábor
Šalingová, Anna
Skalická, Katarína
Brennerová, Katarína
Jančová, Emília
Baráth, Peter
Mucha, Ján
Nemčovič, Marek
author_facet Kodríková, Rebeka
Pakanová, Zuzana
Krchňák, Maroš
Šedivá, Mária
Šesták, Sergej
Květoň, Filip
Beke, Gábor
Šalingová, Anna
Skalická, Katarína
Brennerová, Katarína
Jančová, Emília
Baráth, Peter
Mucha, Ján
Nemčovič, Marek
author_sort Kodríková, Rebeka
collection PubMed
description Congenital disorders of glycosylation (CDG) are a group of rare inherited metabolic disorders caused by a defect in the process of protein glycosylation. In this work, we present a comprehensive glycoprofile analysis of a male patient with a novel missense variant in the SLC35A2 gene, coding a galactose transporter that translocates UDP-galactose from the cytosol to the lumen of the endoplasmic reticulum and Golgi apparatus. Isoelectric focusing of serum transferrin, which resulted in a CDG type II pattern, was followed by structural analysis of transferrin and serum N-glycans, as well as the analysis of apolipoprotein CIII O-glycans by mass spectrometry. An abnormal serum N-glycoprofile with significantly increased levels of agalactosylated (Hex3HexNAc4-5 and Hex3HexNAc5Fuc1) and monogalactosylated (Hex4HexNAc4 ± NeuAc1) N-glycans was observed. Additionally, whole exome sequencing and Sanger sequencing revealed de novo hemizygous c.461T > C (p.Leu154Pro) mutation in the SLC35A2 gene. Based on the combination of biochemical, analytical, and genomic approaches, the set of distinctive N-glycan biomarkers was characterized. Potentially, the set of identified aberrant N-glycans can be specific for other variants causing SLC35A2-CDG and can distinguish this disorder from the other CDGs or other defects in the galactose metabolism.
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spelling pubmed-99529022023-02-25 N-Glycoprofiling of SLC35A2-CDG: Patient with a Novel Hemizygous Variant Kodríková, Rebeka Pakanová, Zuzana Krchňák, Maroš Šedivá, Mária Šesták, Sergej Květoň, Filip Beke, Gábor Šalingová, Anna Skalická, Katarína Brennerová, Katarína Jančová, Emília Baráth, Peter Mucha, Ján Nemčovič, Marek Biomedicines Article Congenital disorders of glycosylation (CDG) are a group of rare inherited metabolic disorders caused by a defect in the process of protein glycosylation. In this work, we present a comprehensive glycoprofile analysis of a male patient with a novel missense variant in the SLC35A2 gene, coding a galactose transporter that translocates UDP-galactose from the cytosol to the lumen of the endoplasmic reticulum and Golgi apparatus. Isoelectric focusing of serum transferrin, which resulted in a CDG type II pattern, was followed by structural analysis of transferrin and serum N-glycans, as well as the analysis of apolipoprotein CIII O-glycans by mass spectrometry. An abnormal serum N-glycoprofile with significantly increased levels of agalactosylated (Hex3HexNAc4-5 and Hex3HexNAc5Fuc1) and monogalactosylated (Hex4HexNAc4 ± NeuAc1) N-glycans was observed. Additionally, whole exome sequencing and Sanger sequencing revealed de novo hemizygous c.461T > C (p.Leu154Pro) mutation in the SLC35A2 gene. Based on the combination of biochemical, analytical, and genomic approaches, the set of distinctive N-glycan biomarkers was characterized. Potentially, the set of identified aberrant N-glycans can be specific for other variants causing SLC35A2-CDG and can distinguish this disorder from the other CDGs or other defects in the galactose metabolism. MDPI 2023-02-16 /pmc/articles/PMC9952902/ /pubmed/36831116 http://dx.doi.org/10.3390/biomedicines11020580 Text en © 2023 by the authors. https://creativecommons.org/licenses/by/4.0/Licensee MDPI, Basel, Switzerland. This article is an open access article distributed under the terms and conditions of the Creative Commons Attribution (CC BY) license (https://creativecommons.org/licenses/by/4.0/).
spellingShingle Article
Kodríková, Rebeka
Pakanová, Zuzana
Krchňák, Maroš
Šedivá, Mária
Šesták, Sergej
Květoň, Filip
Beke, Gábor
Šalingová, Anna
Skalická, Katarína
Brennerová, Katarína
Jančová, Emília
Baráth, Peter
Mucha, Ján
Nemčovič, Marek
N-Glycoprofiling of SLC35A2-CDG: Patient with a Novel Hemizygous Variant
title N-Glycoprofiling of SLC35A2-CDG: Patient with a Novel Hemizygous Variant
title_full N-Glycoprofiling of SLC35A2-CDG: Patient with a Novel Hemizygous Variant
title_fullStr N-Glycoprofiling of SLC35A2-CDG: Patient with a Novel Hemizygous Variant
title_full_unstemmed N-Glycoprofiling of SLC35A2-CDG: Patient with a Novel Hemizygous Variant
title_short N-Glycoprofiling of SLC35A2-CDG: Patient with a Novel Hemizygous Variant
title_sort n-glycoprofiling of slc35a2-cdg: patient with a novel hemizygous variant
topic Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9952902/
https://www.ncbi.nlm.nih.gov/pubmed/36831116
http://dx.doi.org/10.3390/biomedicines11020580
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