Inhibition of transglutaminase 2 mitigates transcriptional dysregulation in models of Huntington disease

Caused by a polyglutamine expansion in the huntingtin protein, Huntington's disease leads to striatal degeneration via the transcriptional dysregulation of a number of genes, including those involved in mitochondrial biogenesis. Here we show that transglutaminase 2, which is upregulated in HD,...

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التفاصيل البيبلوغرافية
المؤلف الرئيسي: Sleiman, Sama F. (author)
مؤلفون آخرون: Mashishi, Lata (author), Coppola, Giovanni (author), Thompson, Leslie M. (author), Ratan, Rajiv R. (author), Starkov, Anatoly (author), Antonyak, Marc A. (author)
التنسيق: article
منشور في: 2010
الوصول للمادة أونلاين:http://hdl.handle.net/10725/6142
http://dx.doi.org/10.1002/emmm.201000084
http://libraries.lau.edu.lb/research/laur/terms-of-use/articles.php
http://embomolmed.embopress.org/content/2/9/349.short
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author Sleiman, Sama F.
author2 Mashishi, Lata
Coppola, Giovanni
Thompson, Leslie M.
Ratan, Rajiv R.
Starkov, Anatoly
Antonyak, Marc A.
author2_role author
author
author
author
author
author
author_facet Sleiman, Sama F.
Mashishi, Lata
Coppola, Giovanni
Thompson, Leslie M.
Ratan, Rajiv R.
Starkov, Anatoly
Antonyak, Marc A.
author_role author
dc.creator.none.fl_str_mv Sleiman, Sama F.
Mashishi, Lata
Coppola, Giovanni
Thompson, Leslie M.
Ratan, Rajiv R.
Starkov, Anatoly
Antonyak, Marc A.
dc.date.none.fl_str_mv 2010
2017-09-06T08:57:44Z
2017-09-06T08:57:44Z
2017-09-06
dc.identifier.none.fl_str_mv 1757-4684
http://hdl.handle.net/10725/6142
http://dx.doi.org/10.1002/emmm.201000084
McConoughey, S. J., Basso, M., Niatsetskaya, Z. V., Sleiman, S. F., Smirnova, N. A., Langley, B. C., ... & Li, B. (2010). Inhibition of transglutaminase 2 mitigates transcriptional dysregulation in models of Huntington disease. EMBO molecular medicine, 2(9), 349-370.
http://libraries.lau.edu.lb/research/laur/terms-of-use/articles.php
http://embomolmed.embopress.org/content/2/9/349.short
dc.language.none.fl_str_mv en
dc.relation.none.fl_str_mv EMBO Molecular Medecine
dc.rights.*.fl_str_mv info:eu-repo/semantics/openAccess
dc.title.none.fl_str_mv Inhibition of transglutaminase 2 mitigates transcriptional dysregulation in models of Huntington disease
dc.type.none.fl_str_mv Article
info:eu-repo/semantics/publishedVersion
info:eu-repo/semantics/article
description Caused by a polyglutamine expansion in the huntingtin protein, Huntington's disease leads to striatal degeneration via the transcriptional dysregulation of a number of genes, including those involved in mitochondrial biogenesis. Here we show that transglutaminase 2, which is upregulated in HD, exacerbates transcriptional dysregulation by acting as a selective corepressor of nuclear genes; transglutaminase 2 interacts directly with histone H3 in the nucleus. In a cellular model of HD, transglutaminase inhibition de‐repressed two established regulators of mitochondrial function, PGC‐1α and cytochrome c and reversed susceptibility of human HD cells to the mitochondrial toxin, 3‐nitroproprionic acid; however, protection mediated by transglutaminase inhibition was not associated with improved mitochondrial bioenergetics. A gene microarray analysis indicated that transglutaminase inhibition normalized expression of not only mitochondrial genes but also 40% of genes that are dysregulated in HD striatal neurons, including chaperone and histone genes. Moreover, transglutaminase inhibition attenuated degeneration in a Drosophila model of HD and protected mouse HD striatal neurons from excitotoxicity. Altogether these findings demonstrate that selective TG inhibition broadly corrects transcriptional dysregulation in HD and defines a novel HDAC‐independent epigenetic strategy for treating neurodegeneration
eu_rights_str_mv openAccess
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identifier_str_mv 1757-4684
McConoughey, S. J., Basso, M., Niatsetskaya, Z. V., Sleiman, S. F., Smirnova, N. A., Langley, B. C., ... & Li, B. (2010). Inhibition of transglutaminase 2 mitigates transcriptional dysregulation in models of Huntington disease. EMBO molecular medicine, 2(9), 349-370.
language_invalid_str_mv en
network_acronym_str LAURepo
network_name_str Lebanese American University repository
oai_identifier_str oai:laur.lau.edu.lb:10725/6142
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spelling Inhibition of transglutaminase 2 mitigates transcriptional dysregulation in models of Huntington diseaseSleiman, Sama F.Mashishi, LataCoppola, GiovanniThompson, Leslie M.Ratan, Rajiv R.Starkov, AnatolyAntonyak, Marc A.Caused by a polyglutamine expansion in the huntingtin protein, Huntington's disease leads to striatal degeneration via the transcriptional dysregulation of a number of genes, including those involved in mitochondrial biogenesis. Here we show that transglutaminase 2, which is upregulated in HD, exacerbates transcriptional dysregulation by acting as a selective corepressor of nuclear genes; transglutaminase 2 interacts directly with histone H3 in the nucleus. In a cellular model of HD, transglutaminase inhibition de‐repressed two established regulators of mitochondrial function, PGC‐1α and cytochrome c and reversed susceptibility of human HD cells to the mitochondrial toxin, 3‐nitroproprionic acid; however, protection mediated by transglutaminase inhibition was not associated with improved mitochondrial bioenergetics. A gene microarray analysis indicated that transglutaminase inhibition normalized expression of not only mitochondrial genes but also 40% of genes that are dysregulated in HD striatal neurons, including chaperone and histone genes. Moreover, transglutaminase inhibition attenuated degeneration in a Drosophila model of HD and protected mouse HD striatal neurons from excitotoxicity. Altogether these findings demonstrate that selective TG inhibition broadly corrects transcriptional dysregulation in HD and defines a novel HDAC‐independent epigenetic strategy for treating neurodegenerationPublishedN/A2017-09-06T08:57:44Z2017-09-06T08:57:44Z20102017-09-06Articleinfo:eu-repo/semantics/publishedVersioninfo:eu-repo/semantics/article1757-4684http://hdl.handle.net/10725/6142http://dx.doi.org/10.1002/emmm.201000084McConoughey, S. J., Basso, M., Niatsetskaya, Z. V., Sleiman, S. F., Smirnova, N. A., Langley, B. C., ... & Li, B. (2010). Inhibition of transglutaminase 2 mitigates transcriptional dysregulation in models of Huntington disease. EMBO molecular medicine, 2(9), 349-370.http://libraries.lau.edu.lb/research/laur/terms-of-use/articles.phphttp://embomolmed.embopress.org/content/2/9/349.shortenEMBO Molecular Medecineinfo:eu-repo/semantics/openAccessoai:laur.lau.edu.lb:10725/61422021-03-19T10:00:49Z
spellingShingle Inhibition of transglutaminase 2 mitigates transcriptional dysregulation in models of Huntington disease
Sleiman, Sama F.
status_str publishedVersion
title Inhibition of transglutaminase 2 mitigates transcriptional dysregulation in models of Huntington disease
title_full Inhibition of transglutaminase 2 mitigates transcriptional dysregulation in models of Huntington disease
title_fullStr Inhibition of transglutaminase 2 mitigates transcriptional dysregulation in models of Huntington disease
title_full_unstemmed Inhibition of transglutaminase 2 mitigates transcriptional dysregulation in models of Huntington disease
title_short Inhibition of transglutaminase 2 mitigates transcriptional dysregulation in models of Huntington disease
title_sort Inhibition of transglutaminase 2 mitigates transcriptional dysregulation in models of Huntington disease
url http://hdl.handle.net/10725/6142
http://dx.doi.org/10.1002/emmm.201000084
http://libraries.lau.edu.lb/research/laur/terms-of-use/articles.php
http://embomolmed.embopress.org/content/2/9/349.short