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Identification of a Novel Mutation of β-Spectrin in Hereditary Spherocytosis Using Whole Exome Sequencing

Hereditary spherocytosis (HS), the most commonly inherited hemolytic anemia in northern Europeans, comprises a group of diseases whose heterogeneous genetic basis results in a variable clinical presentation. High-throughput genome sequencing methods have made a leading contribution to the recent pro...

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Autores principales: Bogusławska, Dżamila M., Skulski, Michał, Machnicka, Beata, Potoczek, Stanisław, Kraszewski, Sebastian, Kuliczkowski, Kazimierz, Sikorski, Aleksander F.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: MDPI 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8540824/
https://www.ncbi.nlm.nih.gov/pubmed/34681667
http://dx.doi.org/10.3390/ijms222011007
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author Bogusławska, Dżamila M.
Skulski, Michał
Machnicka, Beata
Potoczek, Stanisław
Kraszewski, Sebastian
Kuliczkowski, Kazimierz
Sikorski, Aleksander F.
author_facet Bogusławska, Dżamila M.
Skulski, Michał
Machnicka, Beata
Potoczek, Stanisław
Kraszewski, Sebastian
Kuliczkowski, Kazimierz
Sikorski, Aleksander F.
author_sort Bogusławska, Dżamila M.
collection PubMed
description Hereditary spherocytosis (HS), the most commonly inherited hemolytic anemia in northern Europeans, comprises a group of diseases whose heterogeneous genetic basis results in a variable clinical presentation. High-throughput genome sequencing methods have made a leading contribution to the recent progress in research on and diagnostics of inherited diseases and inspired us to apply whole exome sequencing (WES) to identify potential mutations in HS. The data presented here reveal a novel mutation probably responsible for HS in a single Polish family. Patients with clinical evidence of HS (clinical symptoms, hematological data, and EMA test) were enrolled in the study. The examination of the resulting WES data showed a number of polymorphisms in 71 genes associated with known erythrocyte pathologies (including membranopathies, enzymopathies, and hemoglobinopathies). Only a single SPTB gene variant indicated the possible molecular mechanism of the disease in the studied family. The new missense mutation p.C183Y was identified using WES in the SPTB gene, which is most likely the cause of clinical symptoms typical of hereditary spherocytosis (membranopathy) due to structural and functional impairments of human β-spectrin. This mutation allows for a better understanding of the molecular mechanism(s) of one of the membranopathies, hereditary spherocytosis.
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spelling pubmed-85408242021-10-24 Identification of a Novel Mutation of β-Spectrin in Hereditary Spherocytosis Using Whole Exome Sequencing Bogusławska, Dżamila M. Skulski, Michał Machnicka, Beata Potoczek, Stanisław Kraszewski, Sebastian Kuliczkowski, Kazimierz Sikorski, Aleksander F. Int J Mol Sci Case Report Hereditary spherocytosis (HS), the most commonly inherited hemolytic anemia in northern Europeans, comprises a group of diseases whose heterogeneous genetic basis results in a variable clinical presentation. High-throughput genome sequencing methods have made a leading contribution to the recent progress in research on and diagnostics of inherited diseases and inspired us to apply whole exome sequencing (WES) to identify potential mutations in HS. The data presented here reveal a novel mutation probably responsible for HS in a single Polish family. Patients with clinical evidence of HS (clinical symptoms, hematological data, and EMA test) were enrolled in the study. The examination of the resulting WES data showed a number of polymorphisms in 71 genes associated with known erythrocyte pathologies (including membranopathies, enzymopathies, and hemoglobinopathies). Only a single SPTB gene variant indicated the possible molecular mechanism of the disease in the studied family. The new missense mutation p.C183Y was identified using WES in the SPTB gene, which is most likely the cause of clinical symptoms typical of hereditary spherocytosis (membranopathy) due to structural and functional impairments of human β-spectrin. This mutation allows for a better understanding of the molecular mechanism(s) of one of the membranopathies, hereditary spherocytosis. MDPI 2021-10-12 /pmc/articles/PMC8540824/ /pubmed/34681667 http://dx.doi.org/10.3390/ijms222011007 Text en © 2021 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 Case Report
Bogusławska, Dżamila M.
Skulski, Michał
Machnicka, Beata
Potoczek, Stanisław
Kraszewski, Sebastian
Kuliczkowski, Kazimierz
Sikorski, Aleksander F.
Identification of a Novel Mutation of β-Spectrin in Hereditary Spherocytosis Using Whole Exome Sequencing
title Identification of a Novel Mutation of β-Spectrin in Hereditary Spherocytosis Using Whole Exome Sequencing
title_full Identification of a Novel Mutation of β-Spectrin in Hereditary Spherocytosis Using Whole Exome Sequencing
title_fullStr Identification of a Novel Mutation of β-Spectrin in Hereditary Spherocytosis Using Whole Exome Sequencing
title_full_unstemmed Identification of a Novel Mutation of β-Spectrin in Hereditary Spherocytosis Using Whole Exome Sequencing
title_short Identification of a Novel Mutation of β-Spectrin in Hereditary Spherocytosis Using Whole Exome Sequencing
title_sort identification of a novel mutation of β-spectrin in hereditary spherocytosis using whole exome sequencing
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8540824/
https://www.ncbi.nlm.nih.gov/pubmed/34681667
http://dx.doi.org/10.3390/ijms222011007
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