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Blast crisis Ph+ chronic myeloid leukemia with NUP98/HOXA13 up-regulating MSI2

BACKGROUND: Musashi2(Msi2)-Numb pathway de-regulation is a molecular mechanism underlying the transition of chronic phase Ph + CML to deadly blast crisis, particularly in cases with a NUP98/HOXA9 fusion from a t(7;11)(p15;p15). This study provides new insights on the mechanisms cooperating in drivin...

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Autores principales: Di Giacomo, Danika, Pierini, Valentina, Barba, Gianluca, Ceccarelli, Veronica, Vecchini, Alba, Mecucci, Cristina
Formato: Online Artículo Texto
Lenguaje:English
Publicado: BioMed Central 2014
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4071805/
https://www.ncbi.nlm.nih.gov/pubmed/24971156
http://dx.doi.org/10.1186/1755-8166-7-42
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author Di Giacomo, Danika
Pierini, Valentina
Barba, Gianluca
Ceccarelli, Veronica
Vecchini, Alba
Mecucci, Cristina
author_facet Di Giacomo, Danika
Pierini, Valentina
Barba, Gianluca
Ceccarelli, Veronica
Vecchini, Alba
Mecucci, Cristina
author_sort Di Giacomo, Danika
collection PubMed
description BACKGROUND: Musashi2(Msi2)-Numb pathway de-regulation is a molecular mechanism underlying the transition of chronic phase Ph + CML to deadly blast crisis, particularly in cases with a NUP98/HOXA9 fusion from a t(7;11)(p15;p15). This study provides new insights on the mechanisms cooperating in driving MSI2 over-expression and progression of Ph-positive CML. RESULTS: Herein we describe a t(7;11)(p15;p15) originating a NUP98 fusion with HOXA13, at 7p15, in a 39 year-old man in blast crisis of Ph-positive CML. Both MSI2 and HOXA9 were evaluated by quantitative RT-PCR in our patient and in a series of haematological malignancies. Up-regulation of both genes emerged only in the presence of NUP98/HOXA13 gene fusion. However, over-expression of MSI2, but not HOXA9, was found in 2 cases of Ph + blast crisis with additional chromosome aberrations other than t(7;11). To determine the mechanisms underlying MSI2 over-expression in our patient we performed Chromatin Immunoprecipitation and found that NUP98/HOXA13 fusion protein deregulates MSI2 gene by binding its promoter. CONCLUSIONS: To the best of our knowledge, this is the first molecular characterization of NUP98/HOXA13 fusion in blast crisis of Ph + CML. Our findings suggest cooperative mechanisms of MSI2 over-expression driven by HOXA proteins and strongly supports MSI2 as a prognostic marker and a candidate in target treatment of CML.
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spelling pubmed-40718052014-06-27 Blast crisis Ph+ chronic myeloid leukemia with NUP98/HOXA13 up-regulating MSI2 Di Giacomo, Danika Pierini, Valentina Barba, Gianluca Ceccarelli, Veronica Vecchini, Alba Mecucci, Cristina Mol Cytogenet Case Report BACKGROUND: Musashi2(Msi2)-Numb pathway de-regulation is a molecular mechanism underlying the transition of chronic phase Ph + CML to deadly blast crisis, particularly in cases with a NUP98/HOXA9 fusion from a t(7;11)(p15;p15). This study provides new insights on the mechanisms cooperating in driving MSI2 over-expression and progression of Ph-positive CML. RESULTS: Herein we describe a t(7;11)(p15;p15) originating a NUP98 fusion with HOXA13, at 7p15, in a 39 year-old man in blast crisis of Ph-positive CML. Both MSI2 and HOXA9 were evaluated by quantitative RT-PCR in our patient and in a series of haematological malignancies. Up-regulation of both genes emerged only in the presence of NUP98/HOXA13 gene fusion. However, over-expression of MSI2, but not HOXA9, was found in 2 cases of Ph + blast crisis with additional chromosome aberrations other than t(7;11). To determine the mechanisms underlying MSI2 over-expression in our patient we performed Chromatin Immunoprecipitation and found that NUP98/HOXA13 fusion protein deregulates MSI2 gene by binding its promoter. CONCLUSIONS: To the best of our knowledge, this is the first molecular characterization of NUP98/HOXA13 fusion in blast crisis of Ph + CML. Our findings suggest cooperative mechanisms of MSI2 over-expression driven by HOXA proteins and strongly supports MSI2 as a prognostic marker and a candidate in target treatment of CML. BioMed Central 2014-06-20 /pmc/articles/PMC4071805/ /pubmed/24971156 http://dx.doi.org/10.1186/1755-8166-7-42 Text en Copyright © 2014 Di Giacomo et al.; licensee BioMed Central Ltd. http://creativecommons.org/licenses/by/4.0 This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly credited. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.
spellingShingle Case Report
Di Giacomo, Danika
Pierini, Valentina
Barba, Gianluca
Ceccarelli, Veronica
Vecchini, Alba
Mecucci, Cristina
Blast crisis Ph+ chronic myeloid leukemia with NUP98/HOXA13 up-regulating MSI2
title Blast crisis Ph+ chronic myeloid leukemia with NUP98/HOXA13 up-regulating MSI2
title_full Blast crisis Ph+ chronic myeloid leukemia with NUP98/HOXA13 up-regulating MSI2
title_fullStr Blast crisis Ph+ chronic myeloid leukemia with NUP98/HOXA13 up-regulating MSI2
title_full_unstemmed Blast crisis Ph+ chronic myeloid leukemia with NUP98/HOXA13 up-regulating MSI2
title_short Blast crisis Ph+ chronic myeloid leukemia with NUP98/HOXA13 up-regulating MSI2
title_sort blast crisis ph+ chronic myeloid leukemia with nup98/hoxa13 up-regulating msi2
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC4071805/
https://www.ncbi.nlm.nih.gov/pubmed/24971156
http://dx.doi.org/10.1186/1755-8166-7-42
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