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Toblerone: detecting exon deletion events in cancer using RNA-seq

Cancer is driven by mutations of the genome that can result in the activation of oncogenes or repression of tumour suppressor genes. In acute lymphoblastic leukemia (ALL) focal deletions in IKAROS family zinc finger 1 (IKZF1) result in the loss of zinc-finger DNA-binding domains and a dominant negat...

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Autores principales: Lonsdale, Andrew, Halman, Andreas, Brown, Lauren, Kosasih, Hansen, Ekert, Paul, Oshlack, Alicia
Formato: Online Artículo Texto
Lenguaje:English
Publicado: F1000 Research Limited 2023
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10521068/
https://www.ncbi.nlm.nih.gov/pubmed/37767021
http://dx.doi.org/10.12688/f1000research.129490.1
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author Lonsdale, Andrew
Halman, Andreas
Brown, Lauren
Kosasih, Hansen
Ekert, Paul
Oshlack, Alicia
author_facet Lonsdale, Andrew
Halman, Andreas
Brown, Lauren
Kosasih, Hansen
Ekert, Paul
Oshlack, Alicia
author_sort Lonsdale, Andrew
collection PubMed
description Cancer is driven by mutations of the genome that can result in the activation of oncogenes or repression of tumour suppressor genes. In acute lymphoblastic leukemia (ALL) focal deletions in IKAROS family zinc finger 1 (IKZF1) result in the loss of zinc-finger DNA-binding domains and a dominant negative isoform that is associated with higher rates of relapse and  poorer patient outcomes. Clinically, the presence of IKZF1 deletions informs prognosis and treatment options. In this work we developed a method for detecting exon deletions in genes using RNA-seq with application to IKZF1. We developed a pipeline that first uses a custom transcriptome reference consisting of transcripts with exon deletions.  Next, RNA-seq reads are mapped using a pseudoalignment algorithm to identify reads that uniquely support deletions. These are then evaluated for evidence of the deletion with respect to gene expression and other samples. We applied the algorithm, named Toblerone, to a cohort of 99 B-ALL paediatric samples including validated IKZF1 deletions. Furthermore, we developed a graphical desktop app for non-bioinformatics users that can quickly and easily identify and report deletions in IKZF1 from RNA-seq data with informative graphical outputs.
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spelling pubmed-105210682023-09-27 Toblerone: detecting exon deletion events in cancer using RNA-seq Lonsdale, Andrew Halman, Andreas Brown, Lauren Kosasih, Hansen Ekert, Paul Oshlack, Alicia F1000Res Research Article Cancer is driven by mutations of the genome that can result in the activation of oncogenes or repression of tumour suppressor genes. In acute lymphoblastic leukemia (ALL) focal deletions in IKAROS family zinc finger 1 (IKZF1) result in the loss of zinc-finger DNA-binding domains and a dominant negative isoform that is associated with higher rates of relapse and  poorer patient outcomes. Clinically, the presence of IKZF1 deletions informs prognosis and treatment options. In this work we developed a method for detecting exon deletions in genes using RNA-seq with application to IKZF1. We developed a pipeline that first uses a custom transcriptome reference consisting of transcripts with exon deletions.  Next, RNA-seq reads are mapped using a pseudoalignment algorithm to identify reads that uniquely support deletions. These are then evaluated for evidence of the deletion with respect to gene expression and other samples. We applied the algorithm, named Toblerone, to a cohort of 99 B-ALL paediatric samples including validated IKZF1 deletions. Furthermore, we developed a graphical desktop app for non-bioinformatics users that can quickly and easily identify and report deletions in IKZF1 from RNA-seq data with informative graphical outputs. F1000 Research Limited 2023-02-03 /pmc/articles/PMC10521068/ /pubmed/37767021 http://dx.doi.org/10.12688/f1000research.129490.1 Text en Copyright: © 2023 Lonsdale A et al. https://creativecommons.org/licenses/by/4.0/This is an open access article distributed under the terms of the Creative Commons Attribution Licence, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.
spellingShingle Research Article
Lonsdale, Andrew
Halman, Andreas
Brown, Lauren
Kosasih, Hansen
Ekert, Paul
Oshlack, Alicia
Toblerone: detecting exon deletion events in cancer using RNA-seq
title Toblerone: detecting exon deletion events in cancer using RNA-seq
title_full Toblerone: detecting exon deletion events in cancer using RNA-seq
title_fullStr Toblerone: detecting exon deletion events in cancer using RNA-seq
title_full_unstemmed Toblerone: detecting exon deletion events in cancer using RNA-seq
title_short Toblerone: detecting exon deletion events in cancer using RNA-seq
title_sort toblerone: detecting exon deletion events in cancer using rna-seq
topic Research Article
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC10521068/
https://www.ncbi.nlm.nih.gov/pubmed/37767021
http://dx.doi.org/10.12688/f1000research.129490.1
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