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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...
Autores principales: | , , , , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
MDPI
2023
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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. |
format | Online Article Text |
id | pubmed-9952902 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2023 |
publisher | MDPI |
record_format | MEDLINE/PubMed |
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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