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TP53 Abnormalities are underlying the poor outcome associated with chromothripsis in chronic lymphocytic Leukemia patients with complex karyotype

TP53 Abnormalities are underlying the poor outcome associated with chromothripsis in chronic lymphocytic Leukemia patients with complex karyotype
TP53 Abnormalities are underlying the poor outcome associated with chromothripsis in chronic lymphocytic Leukemia patients with complex karyotype
Chromothripsis (cth) has been associated with a dismal outcome and poor prognosis factors in patients with chronic lymphocytic leukemia (CLL). Despite being correlated with high genome instability, previous studies have not assessed the role of cth in the context of genomic complexity. Herein, we analyzed a cohort of 33 CLL patients with cth and compared them against a cohort of 129 non-cth cases with complex karyotypes. Nine cth cases were analyzed using optical genome mapping (OGM). Patterns detected by genomic microarrays were compared and the prognostic value of cth was analyzed. Cth was distributed throughout the genome, with chromosomes 3, 6 and 13 being those most frequently affected. OGM detected 88.1% of the previously known copy number alterations and several additional cth-related rearrangements (median: 9, range: 3-26). Two patterns were identified: one with rearrangements clustered in the region with cth (3/9) and the other involving both chromothriptic and non-chromothriptic chromosomes (6/9). Cases with cth showed a shorter time to first treatment (TTFT) than non-cth patients (median TTFT: 2 m vs. 15 m; p = 0.013). However, when stratifying patients based on TP53 status, cth did not affect TTFT. Only TP53 maintained its significance in the multivariate analysis for TTFT, including cth and genome complexity defined by genomic microarrays (HR: 1.60; p = 0.029). Our findings suggest that TP53 abnormalities, rather than cth itself, underlie the poor prognosis observed in this subset.
TP53, chromothripsis, chronic lymphocytic leukemia, genomic complexity, genomic microarrays, optical genome mapping
2072-6694
Ramos-campoy, Silvia
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Puiggros, Anna
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Kamaso, Joanna
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Beà, Sílvia
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Bougeon, Sandrine
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Larráyoz, María José
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Costa, Dolors
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Parker, Helen
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Rigolin, Gian Matteo
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Blanco, María Laura
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Collado, Rosa
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Ancín, Idoya
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Salgado, Rocío
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Moro-García, Marco A.
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Baumann, Tycho
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Gimeno, Eva
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Moreno, Carolina
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Salido, Marta
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Calvo, Xavier
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Calasanz, María José
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Cuneo, Antonio
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Nguyen-Khac, Florence
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Oscier, David
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Haferlach, Claudia
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Strefford, Jonathan
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Schoumans, Jacqueline
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Espinet Sola, Blanca
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Ramos-campoy, Silvia
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Puiggros, Anna
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Kamaso, Joanna
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Beà, Sílvia
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Bougeon, Sandrine
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Larráyoz, María José
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Costa, Dolors
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Parker, Helen
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Rigolin, Gian Matteo
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Blanco, María Laura
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Collado, Rosa
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Ancín, Idoya
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Salgado, Rocío
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Moro-García, Marco A.
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Baumann, Tycho
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Gimeno, Eva
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Moreno, Carolina
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Salido, Marta
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Calvo, Xavier
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Calasanz, María José
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Cuneo, Antonio
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Nguyen-Khac, Florence
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Oscier, David
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Haferlach, Claudia
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Strefford, Jonathan
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Schoumans, Jacqueline
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Espinet Sola, Blanca
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Ramos-campoy, Silvia, Puiggros, Anna, Kamaso, Joanna, Beà, Sílvia, Bougeon, Sandrine, Larráyoz, María José, Costa, Dolors, Parker, Helen, Rigolin, Gian Matteo, Blanco, María Laura, Collado, Rosa, Ancín, Idoya, Salgado, Rocío, Moro-García, Marco A., Baumann, Tycho, Gimeno, Eva, Moreno, Carolina, Salido, Marta, Calvo, Xavier, Calasanz, María José, Cuneo, Antonio, Nguyen-Khac, Florence, Oscier, David, Haferlach, Claudia, Strefford, Jonathan, Schoumans, Jacqueline and Espinet Sola, Blanca (2022) TP53 Abnormalities are underlying the poor outcome associated with chromothripsis in chronic lymphocytic Leukemia patients with complex karyotype. Cancers, 14 (15), [3715]. (doi:10.3390/cancers14153715).

Record type: Article

Abstract

Chromothripsis (cth) has been associated with a dismal outcome and poor prognosis factors in patients with chronic lymphocytic leukemia (CLL). Despite being correlated with high genome instability, previous studies have not assessed the role of cth in the context of genomic complexity. Herein, we analyzed a cohort of 33 CLL patients with cth and compared them against a cohort of 129 non-cth cases with complex karyotypes. Nine cth cases were analyzed using optical genome mapping (OGM). Patterns detected by genomic microarrays were compared and the prognostic value of cth was analyzed. Cth was distributed throughout the genome, with chromosomes 3, 6 and 13 being those most frequently affected. OGM detected 88.1% of the previously known copy number alterations and several additional cth-related rearrangements (median: 9, range: 3-26). Two patterns were identified: one with rearrangements clustered in the region with cth (3/9) and the other involving both chromothriptic and non-chromothriptic chromosomes (6/9). Cases with cth showed a shorter time to first treatment (TTFT) than non-cth patients (median TTFT: 2 m vs. 15 m; p = 0.013). However, when stratifying patients based on TP53 status, cth did not affect TTFT. Only TP53 maintained its significance in the multivariate analysis for TTFT, including cth and genome complexity defined by genomic microarrays (HR: 1.60; p = 0.029). Our findings suggest that TP53 abnormalities, rather than cth itself, underlie the poor prognosis observed in this subset.

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cancers-14-03715-v3 - Version of Record
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Published date: 29 July 2022
Additional Information: Funding Information: This work was partly supported by grants from Generalitat de Catalunya (17SGR437), Gilead Sciences Fellowship (GLD17/00282), Ministerio de Universidades of Spain (FPU17/00361), Fundación Española de Hematología y Hemoterapia (FEHH-Janssen), Instituto de Salud Carlos III/FEDER (PT17/0015/0011) and the “Xarxa de Bancs de tumors”, sponsored by Pla Director d’Oncologia de Catalunya (XBTC). Publisher Copyright: © 2022 by the authors.
Keywords: TP53, chromothripsis, chronic lymphocytic leukemia, genomic complexity, genomic microarrays, optical genome mapping

Identifiers

Local EPrints ID: 472522
URI: http://eprints.soton.ac.uk/id/eprint/472522
ISSN: 2072-6694
PURE UUID: f84b1fab-769f-4c95-b66d-d5696189d375
ORCID for Jonathan Strefford: ORCID iD orcid.org/0000-0002-0972-2881

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Date deposited: 07 Dec 2022 17:48
Last modified: 17 Mar 2024 02:59

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Contributors

Author: Silvia Ramos-campoy
Author: Anna Puiggros
Author: Joanna Kamaso
Author: Sílvia Beà
Author: Sandrine Bougeon
Author: María José Larráyoz
Author: Dolors Costa
Author: Helen Parker
Author: Gian Matteo Rigolin
Author: María Laura Blanco
Author: Rosa Collado
Author: Idoya Ancín
Author: Rocío Salgado
Author: Marco A. Moro-García
Author: Tycho Baumann
Author: Eva Gimeno
Author: Carolina Moreno
Author: Marta Salido
Author: Xavier Calvo
Author: María José Calasanz
Author: Antonio Cuneo
Author: Florence Nguyen-Khac
Author: David Oscier
Author: Claudia Haferlach
Author: Jacqueline Schoumans
Author: Blanca Espinet Sola

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