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Effects of Friedreich's ataxia GAA repeats on DNA replication in mammalian cells

Friedreich's ataxia (FRDA) is a common hereditary degenerative neuro-muscular disorder caused by expansions of the (GAA)n repeat in the first intron of the frataxin gene. The expanded repeats from parents frequently undergo further significant length changes as they are passed on to progeny. Ex...

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Autores principales: Chandok, Gurangad S., Patel, Mayank P., Mirkin, Sergei M., Krasilnikova, Maria M.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Oxford University Press 2012
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3351192/
https://www.ncbi.nlm.nih.gov/pubmed/22262734
http://dx.doi.org/10.1093/nar/gks021
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author Chandok, Gurangad S.
Patel, Mayank P.
Mirkin, Sergei M.
Krasilnikova, Maria M.
author_facet Chandok, Gurangad S.
Patel, Mayank P.
Mirkin, Sergei M.
Krasilnikova, Maria M.
author_sort Chandok, Gurangad S.
collection PubMed
description Friedreich's ataxia (FRDA) is a common hereditary degenerative neuro-muscular disorder caused by expansions of the (GAA)n repeat in the first intron of the frataxin gene. The expanded repeats from parents frequently undergo further significant length changes as they are passed on to progeny. Expanded repeats also show an age-dependent instability in somatic cells, albeit on a smaller scale than during intergenerational transmissions. Here we studied the effects of (GAA)n repeats of varying lengths and orientations on the episomal DNA replication in mammalian cells. We have recently shown that the very first round of the transfected DNA replication occurs in the lack of the mature chromatin, does not depend on the episomal replication origin and initiates at multiple single-stranded regions of plasmid DNA. We now found that expanded GAA repeats severely block this first replication round post plasmid transfection, while the subsequent replication cycles are only mildly affected. The fact that GAA repeats affect various replication modes in a different way might shed light on their differential expansions characteristic for FRDA.
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spelling pubmed-33511922012-05-14 Effects of Friedreich's ataxia GAA repeats on DNA replication in mammalian cells Chandok, Gurangad S. Patel, Mayank P. Mirkin, Sergei M. Krasilnikova, Maria M. Nucleic Acids Res Genome Integrity, Repair and Replication Friedreich's ataxia (FRDA) is a common hereditary degenerative neuro-muscular disorder caused by expansions of the (GAA)n repeat in the first intron of the frataxin gene. The expanded repeats from parents frequently undergo further significant length changes as they are passed on to progeny. Expanded repeats also show an age-dependent instability in somatic cells, albeit on a smaller scale than during intergenerational transmissions. Here we studied the effects of (GAA)n repeats of varying lengths and orientations on the episomal DNA replication in mammalian cells. We have recently shown that the very first round of the transfected DNA replication occurs in the lack of the mature chromatin, does not depend on the episomal replication origin and initiates at multiple single-stranded regions of plasmid DNA. We now found that expanded GAA repeats severely block this first replication round post plasmid transfection, while the subsequent replication cycles are only mildly affected. The fact that GAA repeats affect various replication modes in a different way might shed light on their differential expansions characteristic for FRDA. Oxford University Press 2012-05 2012-01-19 /pmc/articles/PMC3351192/ /pubmed/22262734 http://dx.doi.org/10.1093/nar/gks021 Text en © The Author(s) 2012. Published by Oxford University Press. http://creativecommons.org/licenses/by-nc/3.0 This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/3.0), which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Genome Integrity, Repair and Replication
Chandok, Gurangad S.
Patel, Mayank P.
Mirkin, Sergei M.
Krasilnikova, Maria M.
Effects of Friedreich's ataxia GAA repeats on DNA replication in mammalian cells
title Effects of Friedreich's ataxia GAA repeats on DNA replication in mammalian cells
title_full Effects of Friedreich's ataxia GAA repeats on DNA replication in mammalian cells
title_fullStr Effects of Friedreich's ataxia GAA repeats on DNA replication in mammalian cells
title_full_unstemmed Effects of Friedreich's ataxia GAA repeats on DNA replication in mammalian cells
title_short Effects of Friedreich's ataxia GAA repeats on DNA replication in mammalian cells
title_sort effects of friedreich's ataxia gaa repeats on dna replication in mammalian cells
topic Genome Integrity, Repair and Replication
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC3351192/
https://www.ncbi.nlm.nih.gov/pubmed/22262734
http://dx.doi.org/10.1093/nar/gks021
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