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Exploring Neuronal Vulnerability to Head Trauma Using a Whole Exome Approach
Brain injuries are associated with oxidative stress and a need to restore neuronal homeostasis. Mutations in ion channel genes, in particular CACNA1A, have been implicated in familial hemiplegic migraine (FHM) and in the development of concussion-related symptoms in response to trivial head trauma....
Autores principales: | , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Mary Ann Liebert, Inc., publishers
2020
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7462038/ https://www.ncbi.nlm.nih.gov/pubmed/32233732 http://dx.doi.org/10.1089/neu.2019.6962 |
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author | Ibrahim, Omar Sutherland, Heidi G. Maksemous, Neven Smith, Robert Haupt, Larisa M. Griffiths, Lyn R. |
author_facet | Ibrahim, Omar Sutherland, Heidi G. Maksemous, Neven Smith, Robert Haupt, Larisa M. Griffiths, Lyn R. |
author_sort | Ibrahim, Omar |
collection | PubMed |
description | Brain injuries are associated with oxidative stress and a need to restore neuronal homeostasis. Mutations in ion channel genes, in particular CACNA1A, have been implicated in familial hemiplegic migraine (FHM) and in the development of concussion-related symptoms in response to trivial head trauma. The aim of this study was to explore the potential role of variants in other ion channel genes in the development of such responses. We conducted whole exome sequencing (WES) on16 individuals who developed a range of neurological and concussion-related symptoms following minor or trivial head injuries. All individuals were initially tested and shown to be negative for mutations in known FHM genes. Variants identified from the WES results were filtered to identify rare variants (minor allele frequency [MAF] <0.01) in genes related to neural processes as well as genes highly expressed in the brain using a combination of in silico prediction tools (SIFT, PolyPhen, PredictSNP, Mutation Taster, and Mutation Assessor). Rare (MAF <0.001) or novel heterozygous variants in 7 ion channel genes were identified in 37.5% (6/16) of the cases (CACNA1I, CACNA1C, ATP10A, ATP7B, KCNAB1, KCNJ10, and SLC26A4), rare variants in neurotransmitter genes were found in 2 cases (GABRG1 and GRIK1), and rare variants in 3 ubiquitin-related genes identified in 4 cases (SQSTM1, TRIM2, and HECTD1). In this study, the largest proportion of potentially pathogenic variants in individuals with severe responses to minor head trauma were identified in genes previously implicated in migraine and seizure-related autosomal recessive neurological disorders. Together with results implicating variants in the hemiplegic migraine genes, CACNA1A and ATP1A2, in severe head trauma response, our results support a role for heterozygous deleterious mutations in genes implicated in neurological dysfunction and potentially increasing the risk of poor response to trivial head trauma. |
format | Online Article Text |
id | pubmed-7462038 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2020 |
publisher | Mary Ann Liebert, Inc., publishers |
record_format | MEDLINE/PubMed |
spelling | pubmed-74620382020-09-01 Exploring Neuronal Vulnerability to Head Trauma Using a Whole Exome Approach Ibrahim, Omar Sutherland, Heidi G. Maksemous, Neven Smith, Robert Haupt, Larisa M. Griffiths, Lyn R. J Neurotrauma Original Articles Brain injuries are associated with oxidative stress and a need to restore neuronal homeostasis. Mutations in ion channel genes, in particular CACNA1A, have been implicated in familial hemiplegic migraine (FHM) and in the development of concussion-related symptoms in response to trivial head trauma. The aim of this study was to explore the potential role of variants in other ion channel genes in the development of such responses. We conducted whole exome sequencing (WES) on16 individuals who developed a range of neurological and concussion-related symptoms following minor or trivial head injuries. All individuals were initially tested and shown to be negative for mutations in known FHM genes. Variants identified from the WES results were filtered to identify rare variants (minor allele frequency [MAF] <0.01) in genes related to neural processes as well as genes highly expressed in the brain using a combination of in silico prediction tools (SIFT, PolyPhen, PredictSNP, Mutation Taster, and Mutation Assessor). Rare (MAF <0.001) or novel heterozygous variants in 7 ion channel genes were identified in 37.5% (6/16) of the cases (CACNA1I, CACNA1C, ATP10A, ATP7B, KCNAB1, KCNJ10, and SLC26A4), rare variants in neurotransmitter genes were found in 2 cases (GABRG1 and GRIK1), and rare variants in 3 ubiquitin-related genes identified in 4 cases (SQSTM1, TRIM2, and HECTD1). In this study, the largest proportion of potentially pathogenic variants in individuals with severe responses to minor head trauma were identified in genes previously implicated in migraine and seizure-related autosomal recessive neurological disorders. Together with results implicating variants in the hemiplegic migraine genes, CACNA1A and ATP1A2, in severe head trauma response, our results support a role for heterozygous deleterious mutations in genes implicated in neurological dysfunction and potentially increasing the risk of poor response to trivial head trauma. Mary Ann Liebert, Inc., publishers 2020-09-01 2020-08-14 /pmc/articles/PMC7462038/ /pubmed/32233732 http://dx.doi.org/10.1089/neu.2019.6962 Text en © Omar Ibrahim et al., 2020; Published by Mary Ann Liebert, Inc. This Open Access article is distributed under the terms of the Creative Commons License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. |
spellingShingle | Original Articles Ibrahim, Omar Sutherland, Heidi G. Maksemous, Neven Smith, Robert Haupt, Larisa M. Griffiths, Lyn R. Exploring Neuronal Vulnerability to Head Trauma Using a Whole Exome Approach |
title | Exploring Neuronal Vulnerability to Head Trauma Using a Whole Exome Approach |
title_full | Exploring Neuronal Vulnerability to Head Trauma Using a Whole Exome Approach |
title_fullStr | Exploring Neuronal Vulnerability to Head Trauma Using a Whole Exome Approach |
title_full_unstemmed | Exploring Neuronal Vulnerability to Head Trauma Using a Whole Exome Approach |
title_short | Exploring Neuronal Vulnerability to Head Trauma Using a Whole Exome Approach |
title_sort | exploring neuronal vulnerability to head trauma using a whole exome approach |
topic | Original Articles |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC7462038/ https://www.ncbi.nlm.nih.gov/pubmed/32233732 http://dx.doi.org/10.1089/neu.2019.6962 |
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