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Conformational stabilization as a strategy to prevent nucleophosmin mislocalization in leukemia
Nucleophosmin (NPM) is a nucleolar protein involved in ribosome assembly and cell homeostasis. Mutations in the C-terminal domain of NPM that impair native folding and localization are associated with acute myeloid leukemia (AML). We have performed a high-throughput screening searching for compounds...
Autores principales: | , , , , , , , , , , |
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Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Nature Publishing Group UK
2017
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Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5655693/ https://www.ncbi.nlm.nih.gov/pubmed/29066752 http://dx.doi.org/10.1038/s41598-017-14497-4 |
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author | Urbaneja, María A. Skjærven, Lars Aubi, Oscar Underhaug, Jarl López, David J. Arregi, Igor Alonso-Mariño, Marián Cuevas, Andoni Rodríguez, José A. Martinez, Aurora Bañuelos, Sonia |
author_facet | Urbaneja, María A. Skjærven, Lars Aubi, Oscar Underhaug, Jarl López, David J. Arregi, Igor Alonso-Mariño, Marián Cuevas, Andoni Rodríguez, José A. Martinez, Aurora Bañuelos, Sonia |
author_sort | Urbaneja, María A. |
collection | PubMed |
description | Nucleophosmin (NPM) is a nucleolar protein involved in ribosome assembly and cell homeostasis. Mutations in the C-terminal domain of NPM that impair native folding and localization are associated with acute myeloid leukemia (AML). We have performed a high-throughput screening searching for compounds that stabilize the C-terminal domain. We identified three hit compounds which show the ability to increase the thermal stability of both the C-terminal domain as well as full-length NPM. The best hit also seemed to favor folding of an AML-like mutant. Computational pocket identification and molecular docking support a stabilization mechanism based on binding of the phenyl/benzene group of the compounds to a particular hydrophobic pocket and additional polar interactions with solvent-accessible residues. Since these results indicate a chaperoning potential of our candidate hits, we tested their effect on the subcellular localization of AML-like mutants. Two compounds partially alleviated the aggregation and restored nucleolar localization of misfolded mutants. The identified hits appear promising as pharmacological chaperones aimed at therapies for AML based on conformational stabilization of NPM. |
format | Online Article Text |
id | pubmed-5655693 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2017 |
publisher | Nature Publishing Group UK |
record_format | MEDLINE/PubMed |
spelling | pubmed-56556932017-10-31 Conformational stabilization as a strategy to prevent nucleophosmin mislocalization in leukemia Urbaneja, María A. Skjærven, Lars Aubi, Oscar Underhaug, Jarl López, David J. Arregi, Igor Alonso-Mariño, Marián Cuevas, Andoni Rodríguez, José A. Martinez, Aurora Bañuelos, Sonia Sci Rep Article Nucleophosmin (NPM) is a nucleolar protein involved in ribosome assembly and cell homeostasis. Mutations in the C-terminal domain of NPM that impair native folding and localization are associated with acute myeloid leukemia (AML). We have performed a high-throughput screening searching for compounds that stabilize the C-terminal domain. We identified three hit compounds which show the ability to increase the thermal stability of both the C-terminal domain as well as full-length NPM. The best hit also seemed to favor folding of an AML-like mutant. Computational pocket identification and molecular docking support a stabilization mechanism based on binding of the phenyl/benzene group of the compounds to a particular hydrophobic pocket and additional polar interactions with solvent-accessible residues. Since these results indicate a chaperoning potential of our candidate hits, we tested their effect on the subcellular localization of AML-like mutants. Two compounds partially alleviated the aggregation and restored nucleolar localization of misfolded mutants. The identified hits appear promising as pharmacological chaperones aimed at therapies for AML based on conformational stabilization of NPM. Nature Publishing Group UK 2017-10-24 /pmc/articles/PMC5655693/ /pubmed/29066752 http://dx.doi.org/10.1038/s41598-017-14497-4 Text en © The Author(s) 2017 Open Access This 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 license, and indicate if changes were made. The images or other third party material in this article are included in the article’s Creative Commons license, unless indicated otherwise in a credit line to the material. If material is not included in the article’s Creative Commons license 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 license, visit http://creativecommons.org/licenses/by/4.0/. |
spellingShingle | Article Urbaneja, María A. Skjærven, Lars Aubi, Oscar Underhaug, Jarl López, David J. Arregi, Igor Alonso-Mariño, Marián Cuevas, Andoni Rodríguez, José A. Martinez, Aurora Bañuelos, Sonia Conformational stabilization as a strategy to prevent nucleophosmin mislocalization in leukemia |
title | Conformational stabilization as a strategy to prevent nucleophosmin mislocalization in leukemia |
title_full | Conformational stabilization as a strategy to prevent nucleophosmin mislocalization in leukemia |
title_fullStr | Conformational stabilization as a strategy to prevent nucleophosmin mislocalization in leukemia |
title_full_unstemmed | Conformational stabilization as a strategy to prevent nucleophosmin mislocalization in leukemia |
title_short | Conformational stabilization as a strategy to prevent nucleophosmin mislocalization in leukemia |
title_sort | conformational stabilization as a strategy to prevent nucleophosmin mislocalization in leukemia |
topic | Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5655693/ https://www.ncbi.nlm.nih.gov/pubmed/29066752 http://dx.doi.org/10.1038/s41598-017-14497-4 |
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