A convergent malignant phenotype in B-cell acute lymphoblastic leukemia involving the splicing factor SRRM1

A significant proportion of infant B-cell acute lymphoblastic leukemia (B-ALL) patients remains with a dismal prognosis due to yet undetermined mechanisms. We performed a comprehensive multicohort analysis of gene expression, gene fusions, and RNA splicing alterations to uncover molecular signatures...

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Autores: Closa, Adrià, Reixachs i Solé, Marina, Fuentes-Fayos, Antonio C., Hayer, Katharina E., Melero, Juan L., Adriaanse, Fabienne R.S., Bos, Romy S., Torres-Diz, Manuel, Hunger, Stephen P., Roberts, Kathryn G., Mullighan, Charles G., Stam, Ronald W., Thomas-Tikhonenko, Andrei, Castaño, Justo P., Luque, Raul M., Eyras Jiménez, Eduardo
Tipo de recurso: artículo
Estado:Versión publicada
Fecha de publicación:2022
País:España
Institución:Varias* (Consorci de Biblioteques Universitáries de Catalunya, Centre de Serveis Científics i Acadèmics de Catalunya)
Repositorio:Recercat. Dipósit de la Recerca de Catalunya
OAI Identifier:oai:recercat.cat:10230/56499
Acceso en línea:http://hdl.handle.net/10230/56499
http://dx.doi.org/10.1093/narcan/zcac041
Access Level:acceso abierto
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spelling A convergent malignant phenotype in B-cell acute lymphoblastic leukemia involving the splicing factor SRRM1Closa, AdriàReixachs i Solé, MarinaFuentes-Fayos, Antonio C.Hayer, Katharina E.Melero, Juan L.Adriaanse, Fabienne R.S.Bos, Romy S.Torres-Diz, ManuelHunger, Stephen P.Roberts, Kathryn G.Mullighan, Charles G.Stam, Ronald W.Thomas-Tikhonenko, AndreiCastaño, Justo P.Luque, Raul M.Eyras Jiménez, EduardoA significant proportion of infant B-cell acute lymphoblastic leukemia (B-ALL) patients remains with a dismal prognosis due to yet undetermined mechanisms. We performed a comprehensive multicohort analysis of gene expression, gene fusions, and RNA splicing alterations to uncover molecular signatures potentially linked to the observed poor outcome. We identified 87 fusions with significant allele frequency across patients and shared functional impacts, suggesting common mechanisms across fusions. We further identified a gene expression signature that predicts high risk independently of the gene fusion background and includes the upregulation of the splicing factor SRRM1. Experiments in B-ALL cell lines provided further evidence for the role of SRRM1 on cell survival, proliferation, and invasion. Supplementary analysis revealed that SRRM1 potentially modulates splicing events associated with poor outcomes through protein-protein interactions with other splicing factors. Our findings reveal a potential convergent mechanism of aberrant RNA processing that sustains a malignant phenotype independently of the underlying gene fusion and that could potentially complement current clinical strategies in infant B-ALL.Spanish Ministerio de Ciencia, Innovación y Universidades [BIO2017-85364-R]; EMBL Australia (to A.C., M.R-S., J.L.M., E.E.); National Institutes of Health [U01 CA232563 to A.T-T.]; M.T-D. acknowledges support from the Ellen Weisberg Fund: Advancing Breakthroughs in Pediatric Cancer. This work was further supported by the Spanish Ministry of Science and Innovation [MICINN, PID2019-105201RB-I00 to J.P.C.]; Junta de Andalucía [BIO-0139]; Universidad de Córdoba-FEDER [UCO-202099901918904 to J.P.C.]; GETNE2019 Research grant (to J.P.C.); CIBERobn Fisiopatología de la Obesidad y Nutrición (CIBER is an initiative of Instituto de Salud Carlos III, co-funded by the European Union: ERDF/ESF, ‘Investing in your future’).Oxford University Press202320232022info:eu-repo/semantics/articleinfo:eu-repo/semantics/publishedVersionapplication/pdfapplication/pdfhttp://hdl.handle.net/10230/56499http://dx.doi.org/10.1093/narcan/zcac041reponame:Recercat. Dipósit de la Recerca de Catalunyainstname:Varias* (Consorci de Biblioteques Universitáries de Catalunya, Centre de Serveis Científics i Acadèmics de Catalunya)InglésNAR Cancer. 2022 Dec 9;4(4):zcac041info:eu-repo/grantAgreement/ES/2PE/BIO2017-85364-Rinfo:eu-repo/grantAgreement/ES/2PE/PID2019-105201RB-I00© The Author(s) 2022. Published by Oxford University Press on behalf of NAR Cancer. This is an Open Access article distributed under the terms of the Creative Commons Attribution License (https://creativecommons.org/licenses/by/4.0/), which permits unrestricted reuse, distribution, and reproduction in any medium, provided the original work is properly cited.http://creativecommons.org/licenses/by/4.0/info:eu-repo/semantics/openAccessoai:recercat.cat:10230/564992026-05-29T05:05:01Z
dc.title.none.fl_str_mv A convergent malignant phenotype in B-cell acute lymphoblastic leukemia involving the splicing factor SRRM1
title A convergent malignant phenotype in B-cell acute lymphoblastic leukemia involving the splicing factor SRRM1
spellingShingle A convergent malignant phenotype in B-cell acute lymphoblastic leukemia involving the splicing factor SRRM1
Closa, Adrià
title_short A convergent malignant phenotype in B-cell acute lymphoblastic leukemia involving the splicing factor SRRM1
title_full A convergent malignant phenotype in B-cell acute lymphoblastic leukemia involving the splicing factor SRRM1
title_fullStr A convergent malignant phenotype in B-cell acute lymphoblastic leukemia involving the splicing factor SRRM1
title_full_unstemmed A convergent malignant phenotype in B-cell acute lymphoblastic leukemia involving the splicing factor SRRM1
title_sort A convergent malignant phenotype in B-cell acute lymphoblastic leukemia involving the splicing factor SRRM1
dc.creator.none.fl_str_mv Closa, Adrià
Reixachs i Solé, Marina
Fuentes-Fayos, Antonio C.
Hayer, Katharina E.
Melero, Juan L.
Adriaanse, Fabienne R.S.
Bos, Romy S.
Torres-Diz, Manuel
Hunger, Stephen P.
Roberts, Kathryn G.
Mullighan, Charles G.
Stam, Ronald W.
Thomas-Tikhonenko, Andrei
Castaño, Justo P.
Luque, Raul M.
Eyras Jiménez, Eduardo
author Closa, Adrià
author_facet Closa, Adrià
Reixachs i Solé, Marina
Fuentes-Fayos, Antonio C.
Hayer, Katharina E.
Melero, Juan L.
Adriaanse, Fabienne R.S.
Bos, Romy S.
Torres-Diz, Manuel
Hunger, Stephen P.
Roberts, Kathryn G.
Mullighan, Charles G.
Stam, Ronald W.
Thomas-Tikhonenko, Andrei
Castaño, Justo P.
Luque, Raul M.
Eyras Jiménez, Eduardo
author_role author
author2 Reixachs i Solé, Marina
Fuentes-Fayos, Antonio C.
Hayer, Katharina E.
Melero, Juan L.
Adriaanse, Fabienne R.S.
Bos, Romy S.
Torres-Diz, Manuel
Hunger, Stephen P.
Roberts, Kathryn G.
Mullighan, Charles G.
Stam, Ronald W.
Thomas-Tikhonenko, Andrei
Castaño, Justo P.
Luque, Raul M.
Eyras Jiménez, Eduardo
author2_role author
author
author
author
author
author
author
author
author
author
author
author
author
author
author
description A significant proportion of infant B-cell acute lymphoblastic leukemia (B-ALL) patients remains with a dismal prognosis due to yet undetermined mechanisms. We performed a comprehensive multicohort analysis of gene expression, gene fusions, and RNA splicing alterations to uncover molecular signatures potentially linked to the observed poor outcome. We identified 87 fusions with significant allele frequency across patients and shared functional impacts, suggesting common mechanisms across fusions. We further identified a gene expression signature that predicts high risk independently of the gene fusion background and includes the upregulation of the splicing factor SRRM1. Experiments in B-ALL cell lines provided further evidence for the role of SRRM1 on cell survival, proliferation, and invasion. Supplementary analysis revealed that SRRM1 potentially modulates splicing events associated with poor outcomes through protein-protein interactions with other splicing factors. Our findings reveal a potential convergent mechanism of aberrant RNA processing that sustains a malignant phenotype independently of the underlying gene fusion and that could potentially complement current clinical strategies in infant B-ALL.
publishDate 2022
dc.date.none.fl_str_mv 2022
2023
2023
dc.type.none.fl_str_mv info:eu-repo/semantics/article
info:eu-repo/semantics/publishedVersion
format article
status_str publishedVersion
dc.identifier.none.fl_str_mv http://hdl.handle.net/10230/56499
http://dx.doi.org/10.1093/narcan/zcac041
url http://hdl.handle.net/10230/56499
http://dx.doi.org/10.1093/narcan/zcac041
dc.language.none.fl_str_mv Inglés
language_invalid_str_mv Inglés
dc.relation.none.fl_str_mv NAR Cancer. 2022 Dec 9;4(4):zcac041
info:eu-repo/grantAgreement/ES/2PE/BIO2017-85364-R
info:eu-repo/grantAgreement/ES/2PE/PID2019-105201RB-I00
dc.rights.none.fl_str_mv http://creativecommons.org/licenses/by/4.0/
info:eu-repo/semantics/openAccess
rights_invalid_str_mv http://creativecommons.org/licenses/by/4.0/
eu_rights_str_mv openAccess
dc.format.none.fl_str_mv application/pdf
application/pdf
dc.publisher.none.fl_str_mv Oxford University Press
publisher.none.fl_str_mv Oxford University Press
dc.source.none.fl_str_mv reponame:Recercat. Dipósit de la Recerca de Catalunya
instname:Varias* (Consorci de Biblioteques Universitáries de Catalunya, Centre de Serveis Científics i Acadèmics de Catalunya)
instname_str Varias* (Consorci de Biblioteques Universitáries de Catalunya, Centre de Serveis Científics i Acadèmics de Catalunya)
reponame_str Recercat. Dipósit de la Recerca de Catalunya
collection Recercat. Dipósit de la Recerca de Catalunya
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