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MN1 Neurodevelopmental Disease-Atypical Phenotype Due to a Novel Frameshift Variant in the MN1 Gene

Background: MN1 C-terminal truncation (MCTT) syndrome is caused by variants in the C-terminal region of MN1, which were first described in 2020. The clinical features of MCTT syndrome includes severe neurodevelopmental and brain abnormalities. We reported on a patient who carried the MN1 variant in...

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Autores principales: Tian, Qi, Shu, Li, Zhang, Pu, Zeng, Ting, Cao, Yang, Xi, Hui, Peng, Ying, Wang, Yaqin, Mao, Xiao, Wang, Hua
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Frontiers Media S.A. 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8716923/
https://www.ncbi.nlm.nih.gov/pubmed/34975401
http://dx.doi.org/10.3389/fnmol.2021.789778
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author Tian, Qi
Shu, Li
Zhang, Pu
Zeng, Ting
Cao, Yang
Xi, Hui
Peng, Ying
Wang, Yaqin
Mao, Xiao
Wang, Hua
author_facet Tian, Qi
Shu, Li
Zhang, Pu
Zeng, Ting
Cao, Yang
Xi, Hui
Peng, Ying
Wang, Yaqin
Mao, Xiao
Wang, Hua
author_sort Tian, Qi
collection PubMed
description Background: MN1 C-terminal truncation (MCTT) syndrome is caused by variants in the C-terminal region of MN1, which were first described in 2020. The clinical features of MCTT syndrome includes severe neurodevelopmental and brain abnormalities. We reported on a patient who carried the MN1 variant in the C-terminal region with mild developmental delay and normal brain magnetic resonance image (MRI). Methods: Detailed clinical information was collected in the pedigree. Whole-exome sequencing (WES) accompanied with Sanger sequencing validation were performed. A functional study based on HEK239T cells was performed. Results: A de novo heterozygous c.3734delT: p.L1245fs variant was detected. HEK239T cells transinfected with the de novo variant showed decreased proliferation, enhanced apoptotic rate, and MN1 nuclear aggregation. Conclusion: Our study expended the clinical and genetic spectrum of MCTT which contributes to the genetic counseling of the MN1 gene.
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spelling pubmed-87169232021-12-31 MN1 Neurodevelopmental Disease-Atypical Phenotype Due to a Novel Frameshift Variant in the MN1 Gene Tian, Qi Shu, Li Zhang, Pu Zeng, Ting Cao, Yang Xi, Hui Peng, Ying Wang, Yaqin Mao, Xiao Wang, Hua Front Mol Neurosci Neuroscience Background: MN1 C-terminal truncation (MCTT) syndrome is caused by variants in the C-terminal region of MN1, which were first described in 2020. The clinical features of MCTT syndrome includes severe neurodevelopmental and brain abnormalities. We reported on a patient who carried the MN1 variant in the C-terminal region with mild developmental delay and normal brain magnetic resonance image (MRI). Methods: Detailed clinical information was collected in the pedigree. Whole-exome sequencing (WES) accompanied with Sanger sequencing validation were performed. A functional study based on HEK239T cells was performed. Results: A de novo heterozygous c.3734delT: p.L1245fs variant was detected. HEK239T cells transinfected with the de novo variant showed decreased proliferation, enhanced apoptotic rate, and MN1 nuclear aggregation. Conclusion: Our study expended the clinical and genetic spectrum of MCTT which contributes to the genetic counseling of the MN1 gene. Frontiers Media S.A. 2021-12-16 /pmc/articles/PMC8716923/ /pubmed/34975401 http://dx.doi.org/10.3389/fnmol.2021.789778 Text en Copyright © 2021 Tian, Shu, Zhang, Zeng, Cao, Xi, Peng, Wang, Mao and Wang. https://creativecommons.org/licenses/by/4.0/This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) and the copyright owner(s) are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.
spellingShingle Neuroscience
Tian, Qi
Shu, Li
Zhang, Pu
Zeng, Ting
Cao, Yang
Xi, Hui
Peng, Ying
Wang, Yaqin
Mao, Xiao
Wang, Hua
MN1 Neurodevelopmental Disease-Atypical Phenotype Due to a Novel Frameshift Variant in the MN1 Gene
title MN1 Neurodevelopmental Disease-Atypical Phenotype Due to a Novel Frameshift Variant in the MN1 Gene
title_full MN1 Neurodevelopmental Disease-Atypical Phenotype Due to a Novel Frameshift Variant in the MN1 Gene
title_fullStr MN1 Neurodevelopmental Disease-Atypical Phenotype Due to a Novel Frameshift Variant in the MN1 Gene
title_full_unstemmed MN1 Neurodevelopmental Disease-Atypical Phenotype Due to a Novel Frameshift Variant in the MN1 Gene
title_short MN1 Neurodevelopmental Disease-Atypical Phenotype Due to a Novel Frameshift Variant in the MN1 Gene
title_sort mn1 neurodevelopmental disease-atypical phenotype due to a novel frameshift variant in the mn1 gene
topic Neuroscience
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8716923/
https://www.ncbi.nlm.nih.gov/pubmed/34975401
http://dx.doi.org/10.3389/fnmol.2021.789778
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