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A case of Aicardi-Goutières syndrome caused by TREX1 gene mutation

Aicardi-Goutières syndrome (AGS) is a rare genetic disorder involving the central nervous system and autoimmune abnormalities, leading to severe intellectual and physical disability with poor prognosis. AGS has a phenotype similar to intrauterine viral infection, which often leads to delays in genet...

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Autores principales: Chenhan, Zheng, Jun, Shao, Yang, Ding, Linliang, Yin, Xiaowen, Gu, Chunya, Ji, Xuedong, Deng
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9945611/
https://www.ncbi.nlm.nih.gov/pubmed/36814213
http://dx.doi.org/10.1186/s12884-023-05436-5
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author Chenhan, Zheng
Jun, Shao
Yang, Ding
Linliang, Yin
Xiaowen, Gu
Chunya, Ji
Xuedong, Deng
author_facet Chenhan, Zheng
Jun, Shao
Yang, Ding
Linliang, Yin
Xiaowen, Gu
Chunya, Ji
Xuedong, Deng
author_sort Chenhan, Zheng
collection PubMed
description Aicardi-Goutières syndrome (AGS) is a rare genetic disorder involving the central nervous system and autoimmune abnormalities, leading to severe intellectual and physical disability with poor prognosis. AGS has a phenotype similar to intrauterine viral infection, which often leads to delays in genetic counseling. In this study, we report a case with a prenatal diagnosis of AGS. The first fetal ultrasound detected bilateral lateral ventricle cystic structures, and fetal MRI was performed to identify other signs. The right parietal lobe signal showed cerebral white matter abnormalities, and fetal brain development level was lower than that of normal fetuses of the same gestational age. Whole-exome sequencing revealed that the fetus carried the TREX1:NM_033629.6:exon2:c.294dup:p. C99Mfs*3 variant, suggesting that the c.294dup mutation of the TREX1 gene was the pathogenic mutation site, and the final comprehensive diagnosis was AGS1. In this article, we also reviewed the previous literature for possible phenotypes in the fetus and found that microcephaly and intrauterine growth retardation may be the first and most important markers of the intrauterine phenotype of AGS.
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spelling pubmed-99456112023-02-23 A case of Aicardi-Goutières syndrome caused by TREX1 gene mutation Chenhan, Zheng Jun, Shao Yang, Ding Linliang, Yin Xiaowen, Gu Chunya, Ji Xuedong, Deng BMC Pregnancy Childbirth Case Report Aicardi-Goutières syndrome (AGS) is a rare genetic disorder involving the central nervous system and autoimmune abnormalities, leading to severe intellectual and physical disability with poor prognosis. AGS has a phenotype similar to intrauterine viral infection, which often leads to delays in genetic counseling. In this study, we report a case with a prenatal diagnosis of AGS. The first fetal ultrasound detected bilateral lateral ventricle cystic structures, and fetal MRI was performed to identify other signs. The right parietal lobe signal showed cerebral white matter abnormalities, and fetal brain development level was lower than that of normal fetuses of the same gestational age. Whole-exome sequencing revealed that the fetus carried the TREX1:NM_033629.6:exon2:c.294dup:p. C99Mfs*3 variant, suggesting that the c.294dup mutation of the TREX1 gene was the pathogenic mutation site, and the final comprehensive diagnosis was AGS1. In this article, we also reviewed the previous literature for possible phenotypes in the fetus and found that microcephaly and intrauterine growth retardation may be the first and most important markers of the intrauterine phenotype of AGS. BioMed Central 2023-02-22 /pmc/articles/PMC9945611/ /pubmed/36814213 http://dx.doi.org/10.1186/s12884-023-05436-5 Text en © The Author(s) 2023 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/) . The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/ (https://creativecommons.org/publicdomain/zero/1.0/) ) applies to the data made available in this article, unless otherwise stated in a credit line to the data.
spellingShingle Case Report
Chenhan, Zheng
Jun, Shao
Yang, Ding
Linliang, Yin
Xiaowen, Gu
Chunya, Ji
Xuedong, Deng
A case of Aicardi-Goutières syndrome caused by TREX1 gene mutation
title A case of Aicardi-Goutières syndrome caused by TREX1 gene mutation
title_full A case of Aicardi-Goutières syndrome caused by TREX1 gene mutation
title_fullStr A case of Aicardi-Goutières syndrome caused by TREX1 gene mutation
title_full_unstemmed A case of Aicardi-Goutières syndrome caused by TREX1 gene mutation
title_short A case of Aicardi-Goutières syndrome caused by TREX1 gene mutation
title_sort case of aicardi-goutières syndrome caused by trex1 gene mutation
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9945611/
https://www.ncbi.nlm.nih.gov/pubmed/36814213
http://dx.doi.org/10.1186/s12884-023-05436-5
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