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Whole Genome Sequencing in an Acrodermatitis Enteropathica Family from the Middle East

We report a family from Tabuk, Saudi Arabia, previously screened for Acrodermatitis Enteropathica (AE), in which two siblings presented with typical features of acral dermatitis and a pustular eruption but differing severity. Affected members of our family carry a rare genetic variant, p.Gly512Trp i...

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Autores principales: Abu-Duhier, Faisel, Pooranachandran, Vivetha, McDonagh, Andrew J. G., Messenger, Andrew G., Cooper-Knock, Johnathan, Bakri, Youssef, Heath, Paul R., Tazi-Ahnini, Rachid
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Hindawi 2018
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6106946/
https://www.ncbi.nlm.nih.gov/pubmed/30174688
http://dx.doi.org/10.1155/2018/1284568
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author Abu-Duhier, Faisel
Pooranachandran, Vivetha
McDonagh, Andrew J. G.
Messenger, Andrew G.
Cooper-Knock, Johnathan
Bakri, Youssef
Heath, Paul R.
Tazi-Ahnini, Rachid
author_facet Abu-Duhier, Faisel
Pooranachandran, Vivetha
McDonagh, Andrew J. G.
Messenger, Andrew G.
Cooper-Knock, Johnathan
Bakri, Youssef
Heath, Paul R.
Tazi-Ahnini, Rachid
author_sort Abu-Duhier, Faisel
collection PubMed
description We report a family from Tabuk, Saudi Arabia, previously screened for Acrodermatitis Enteropathica (AE), in which two siblings presented with typical features of acral dermatitis and a pustular eruption but differing severity. Affected members of our family carry a rare genetic variant, p.Gly512Trp in the SLC39A4 gene which encodes a zinc transporter; disease is thought to result from zinc deficiency. Similar mutations have been reported previously; however, the variable severity within cases carrying the p.Gly512Trp variant and in AE overall led us to hypothesise that additional genetic modifiers may be contributing to the disease phenotype. Therefore whole genome sequencing was carried out in five family members, for whom material was available to search for additional modifiers of AE; this included one individual with clinically diagnosed AE. We confirmed that the p.Gly512Trp change in SLC39A4 was the only candidate homozygous change which was sufficiently rare (ExAC allele frequency 1.178e-05) and predicted deleterious (CADD score 35) to be attributable as a fully penetrant cause of AE. To identify other genes which may carry relevant genetic variation, we reviewed the relevant literature and databases including Gene Ontology Consortium, GeneMANIA, GeneCards, and MalaCards to identify zinc transporter genes and possible interacting partners. The affected individual carried variants in RECQL4 and GPAA1 genes with ExAC allele frequency <0.01 and CADD score >10. p.Gly512Trp is highly likely to be the pathogenic variant in this family. This variant was previously detected in a Tunisian proband with perfect genotype-phenotype segregation suggestive of pathogenicity. Further research is required in this area due to small sample size, but attention should be given to RECQL4 and GPAA1 to understand their role in the skin disease.
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spelling pubmed-61069462018-09-02 Whole Genome Sequencing in an Acrodermatitis Enteropathica Family from the Middle East Abu-Duhier, Faisel Pooranachandran, Vivetha McDonagh, Andrew J. G. Messenger, Andrew G. Cooper-Knock, Johnathan Bakri, Youssef Heath, Paul R. Tazi-Ahnini, Rachid Dermatol Res Pract Research Article We report a family from Tabuk, Saudi Arabia, previously screened for Acrodermatitis Enteropathica (AE), in which two siblings presented with typical features of acral dermatitis and a pustular eruption but differing severity. Affected members of our family carry a rare genetic variant, p.Gly512Trp in the SLC39A4 gene which encodes a zinc transporter; disease is thought to result from zinc deficiency. Similar mutations have been reported previously; however, the variable severity within cases carrying the p.Gly512Trp variant and in AE overall led us to hypothesise that additional genetic modifiers may be contributing to the disease phenotype. Therefore whole genome sequencing was carried out in five family members, for whom material was available to search for additional modifiers of AE; this included one individual with clinically diagnosed AE. We confirmed that the p.Gly512Trp change in SLC39A4 was the only candidate homozygous change which was sufficiently rare (ExAC allele frequency 1.178e-05) and predicted deleterious (CADD score 35) to be attributable as a fully penetrant cause of AE. To identify other genes which may carry relevant genetic variation, we reviewed the relevant literature and databases including Gene Ontology Consortium, GeneMANIA, GeneCards, and MalaCards to identify zinc transporter genes and possible interacting partners. The affected individual carried variants in RECQL4 and GPAA1 genes with ExAC allele frequency <0.01 and CADD score >10. p.Gly512Trp is highly likely to be the pathogenic variant in this family. This variant was previously detected in a Tunisian proband with perfect genotype-phenotype segregation suggestive of pathogenicity. Further research is required in this area due to small sample size, but attention should be given to RECQL4 and GPAA1 to understand their role in the skin disease. Hindawi 2018-08-07 /pmc/articles/PMC6106946/ /pubmed/30174688 http://dx.doi.org/10.1155/2018/1284568 Text en Copyright © 2018 Faisel Abu-Duhier et al. https://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Research Article
Abu-Duhier, Faisel
Pooranachandran, Vivetha
McDonagh, Andrew J. G.
Messenger, Andrew G.
Cooper-Knock, Johnathan
Bakri, Youssef
Heath, Paul R.
Tazi-Ahnini, Rachid
Whole Genome Sequencing in an Acrodermatitis Enteropathica Family from the Middle East
title Whole Genome Sequencing in an Acrodermatitis Enteropathica Family from the Middle East
title_full Whole Genome Sequencing in an Acrodermatitis Enteropathica Family from the Middle East
title_fullStr Whole Genome Sequencing in an Acrodermatitis Enteropathica Family from the Middle East
title_full_unstemmed Whole Genome Sequencing in an Acrodermatitis Enteropathica Family from the Middle East
title_short Whole Genome Sequencing in an Acrodermatitis Enteropathica Family from the Middle East
title_sort whole genome sequencing in an acrodermatitis enteropathica family from the middle east
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC6106946/
https://www.ncbi.nlm.nih.gov/pubmed/30174688
http://dx.doi.org/10.1155/2018/1284568
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