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Novel Pathogenic Variant in TGFBR2 Confirmed by Molecular Modeling Is a Rare Cause of Loeys-Dietz Syndrome
Loeys-Dietz syndrome (LDS) is a connective tissue disorder characterized by vascular findings of aneurysm and/or dissection of cerebral, thoracic, or abdominal arteries and skeletal findings. We report a case of a novel pathogenic variant in TGFBR2 and phenotype consistent with classic LDS. The prob...
Autores principales: | , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Hindawi Publishing Corporation
2017
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5253504/ https://www.ncbi.nlm.nih.gov/pubmed/28163941 http://dx.doi.org/10.1155/2017/7263780 |
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author | Zimmermann, Michael T. Urrutia, Raul A. Blackburn, Patrick R. Cousin, Margot A. Boczek, Nicole J. Klee, Eric W. Macmurdo, Colleen Atwal, Paldeep S. |
author_facet | Zimmermann, Michael T. Urrutia, Raul A. Blackburn, Patrick R. Cousin, Margot A. Boczek, Nicole J. Klee, Eric W. Macmurdo, Colleen Atwal, Paldeep S. |
author_sort | Zimmermann, Michael T. |
collection | PubMed |
description | Loeys-Dietz syndrome (LDS) is a connective tissue disorder characterized by vascular findings of aneurysm and/or dissection of cerebral, thoracic, or abdominal arteries and skeletal findings. We report a case of a novel pathogenic variant in TGFBR2 and phenotype consistent with classic LDS. The proband was a 10-year-old presenting to the genetics clinic with an enlarged aortic root (Z-scores 5-6), pectus excavatum, and congenital contractures of the right 2nd and 3rd digit. Molecular testing of TGFBR2 was sent to a commercial laboratory and demonstrated a novel, likely pathogenic, variant in exon 4, c.1061T>C, p.(L354P). Molecular modeling reveals alteration of local protein structure as a result of this pathogenic variant. This pathogenic variant has not been previously reported in LDS and thus expands the pathogenic variant spectrum of this condition. |
format | Online Article Text |
id | pubmed-5253504 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2017 |
publisher | Hindawi Publishing Corporation |
record_format | MEDLINE/PubMed |
spelling | pubmed-52535042017-02-05 Novel Pathogenic Variant in TGFBR2 Confirmed by Molecular Modeling Is a Rare Cause of Loeys-Dietz Syndrome Zimmermann, Michael T. Urrutia, Raul A. Blackburn, Patrick R. Cousin, Margot A. Boczek, Nicole J. Klee, Eric W. Macmurdo, Colleen Atwal, Paldeep S. Case Rep Genet Case Report Loeys-Dietz syndrome (LDS) is a connective tissue disorder characterized by vascular findings of aneurysm and/or dissection of cerebral, thoracic, or abdominal arteries and skeletal findings. We report a case of a novel pathogenic variant in TGFBR2 and phenotype consistent with classic LDS. The proband was a 10-year-old presenting to the genetics clinic with an enlarged aortic root (Z-scores 5-6), pectus excavatum, and congenital contractures of the right 2nd and 3rd digit. Molecular testing of TGFBR2 was sent to a commercial laboratory and demonstrated a novel, likely pathogenic, variant in exon 4, c.1061T>C, p.(L354P). Molecular modeling reveals alteration of local protein structure as a result of this pathogenic variant. This pathogenic variant has not been previously reported in LDS and thus expands the pathogenic variant spectrum of this condition. Hindawi Publishing Corporation 2017 2017-01-09 /pmc/articles/PMC5253504/ /pubmed/28163941 http://dx.doi.org/10.1155/2017/7263780 Text en Copyright © 2017 Michael T. Zimmermann 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 | Case Report Zimmermann, Michael T. Urrutia, Raul A. Blackburn, Patrick R. Cousin, Margot A. Boczek, Nicole J. Klee, Eric W. Macmurdo, Colleen Atwal, Paldeep S. Novel Pathogenic Variant in TGFBR2 Confirmed by Molecular Modeling Is a Rare Cause of Loeys-Dietz Syndrome |
title | Novel Pathogenic Variant in TGFBR2 Confirmed by Molecular Modeling Is a Rare Cause of Loeys-Dietz Syndrome |
title_full | Novel Pathogenic Variant in TGFBR2 Confirmed by Molecular Modeling Is a Rare Cause of Loeys-Dietz Syndrome |
title_fullStr | Novel Pathogenic Variant in TGFBR2 Confirmed by Molecular Modeling Is a Rare Cause of Loeys-Dietz Syndrome |
title_full_unstemmed | Novel Pathogenic Variant in TGFBR2 Confirmed by Molecular Modeling Is a Rare Cause of Loeys-Dietz Syndrome |
title_short | Novel Pathogenic Variant in TGFBR2 Confirmed by Molecular Modeling Is a Rare Cause of Loeys-Dietz Syndrome |
title_sort | novel pathogenic variant in tgfbr2 confirmed by molecular modeling is a rare cause of loeys-dietz syndrome |
topic | Case Report |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5253504/ https://www.ncbi.nlm.nih.gov/pubmed/28163941 http://dx.doi.org/10.1155/2017/7263780 |
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