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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...

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Autores principales: 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
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Nature Publishing Group UK 2017
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.
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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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