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Defining the molecular pathology and consequent phenotypes in Egyptian HB patients

BACKGROUND: Hemophilia B (HB) (also known as Christmas disease) is a rare X-linked recessive disorder characterized by spontaneous or prolonged hemorrhages caused by mutations in Factor 9 (F9) gene leading to deficient or defective coagulation F9. Our study aimed at identifying the causative mutatio...

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Autores principales: El-Kamah, Ghada Y., Mosaad, Rehab M., Taher, Mohamed B., Amr, Khalda S.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Springer Berlin Heidelberg 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8128942/
https://www.ncbi.nlm.nih.gov/pubmed/33999344
http://dx.doi.org/10.1186/s43141-021-00165-8
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author El-Kamah, Ghada Y.
Mosaad, Rehab M.
Taher, Mohamed B.
Amr, Khalda S.
author_facet El-Kamah, Ghada Y.
Mosaad, Rehab M.
Taher, Mohamed B.
Amr, Khalda S.
author_sort El-Kamah, Ghada Y.
collection PubMed
description BACKGROUND: Hemophilia B (HB) (also known as Christmas disease) is a rare X-linked recessive disorder characterized by spontaneous or prolonged hemorrhages caused by mutations in Factor 9 (F9) gene leading to deficient or defective coagulation F9. Our study aimed at identifying the causative mutations within a sample of HB Egyptian patients. The present study comprised clinical data of eleven HB patients descending from six unrelated families and a seventh family including a carrier mother with a history of deceased HB sibling. Sequencing of F9 gene was performed. RESULTS: The study revealed four mutations; two missense NM_000133.3:c.676C>G, (P.Arg226Gly) and NM_000133.3:c.1305T>G, (p.Cys435Trp), and two nonsense mutations NM_000133.3:c.880C>T, (p.Arg294*) and NM_000133.3:c.1150C>T, (p.Arg384*), identified mutations spanned exons 6 and 8 of which a total of three mutations are located in hotspot exon 8 of F9 gene. CONCLUSIONS: Reviewing the literature, this is the first molecular analysis of F9 gene in HB Egyptian patients. Consistent genotype/phenotypic severity correlation could be concluded, helping proper genetic counseling and prenatal decision taking.
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spelling pubmed-81289422021-05-27 Defining the molecular pathology and consequent phenotypes in Egyptian HB patients El-Kamah, Ghada Y. Mosaad, Rehab M. Taher, Mohamed B. Amr, Khalda S. J Genet Eng Biotechnol Research BACKGROUND: Hemophilia B (HB) (also known as Christmas disease) is a rare X-linked recessive disorder characterized by spontaneous or prolonged hemorrhages caused by mutations in Factor 9 (F9) gene leading to deficient or defective coagulation F9. Our study aimed at identifying the causative mutations within a sample of HB Egyptian patients. The present study comprised clinical data of eleven HB patients descending from six unrelated families and a seventh family including a carrier mother with a history of deceased HB sibling. Sequencing of F9 gene was performed. RESULTS: The study revealed four mutations; two missense NM_000133.3:c.676C>G, (P.Arg226Gly) and NM_000133.3:c.1305T>G, (p.Cys435Trp), and two nonsense mutations NM_000133.3:c.880C>T, (p.Arg294*) and NM_000133.3:c.1150C>T, (p.Arg384*), identified mutations spanned exons 6 and 8 of which a total of three mutations are located in hotspot exon 8 of F9 gene. CONCLUSIONS: Reviewing the literature, this is the first molecular analysis of F9 gene in HB Egyptian patients. Consistent genotype/phenotypic severity correlation could be concluded, helping proper genetic counseling and prenatal decision taking. Springer Berlin Heidelberg 2021-05-17 /pmc/articles/PMC8128942/ /pubmed/33999344 http://dx.doi.org/10.1186/s43141-021-00165-8 Text en © The Author(s) 2021 https://creativecommons.org/licenses/by/4.0/Open AccessThis article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/ (https://creativecommons.org/licenses/by/4.0/) .
spellingShingle Research
El-Kamah, Ghada Y.
Mosaad, Rehab M.
Taher, Mohamed B.
Amr, Khalda S.
Defining the molecular pathology and consequent phenotypes in Egyptian HB patients
title Defining the molecular pathology and consequent phenotypes in Egyptian HB patients
title_full Defining the molecular pathology and consequent phenotypes in Egyptian HB patients
title_fullStr Defining the molecular pathology and consequent phenotypes in Egyptian HB patients
title_full_unstemmed Defining the molecular pathology and consequent phenotypes in Egyptian HB patients
title_short Defining the molecular pathology and consequent phenotypes in Egyptian HB patients
title_sort defining the molecular pathology and consequent phenotypes in egyptian hb patients
topic Research
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8128942/
https://www.ncbi.nlm.nih.gov/pubmed/33999344
http://dx.doi.org/10.1186/s43141-021-00165-8
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