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The Prognostic Effect of IKZF1 Deletions in ETV6::RUNX1 and High Hyperdiploid Childhood Acute Lymphoblastic Leukemia

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The Prognostic Effect of IKZF1 Deletions in ETV6::RUNX1 and High Hyperdiploid Childhood Acute Lymphoblastic Leukemia. / Østergaard, Anna; Enshaei, Amir; Pieters, Rob; Vora, Ajay; Horstmann, Martin A; Escherich, Gabriele; Johansson, Bertil; Heyman, Mats; Schmiegelow, Kjeld; Hoogerbrugge, Peter M; den Boer, Monique L; Kuiper, Roland P; Moorman, Anthony V; Boer, Judith M; van Leeuwen, Frank N.

In: HEMASPHERE, Vol. 7, No. 5, 05.2023, p. e875.

Research output: SCORING: Contribution to journal › SCORING: Journal article › Research › peer-review

Harvard

Østergaard, A, Enshaei, A, Pieters, R, Vora, A, Horstmann, MA, Escherich, G, Johansson, B, Heyman, M, Schmiegelow, K, Hoogerbrugge, PM, den Boer, ML, Kuiper, RP, Moorman, AV, Boer, JM & van Leeuwen, FN 2023, 'The Prognostic Effect of IKZF1 Deletions in ETV6::RUNX1 and High Hyperdiploid Childhood Acute Lymphoblastic Leukemia', HEMASPHERE, vol. 7, no. 5, pp. e875. https://doi.org/10.1097/HS9.0000000000000875

APA

Østergaard, A., Enshaei, A., Pieters, R., Vora, A., Horstmann, M. A., Escherich, G., Johansson, B., Heyman, M., Schmiegelow, K., Hoogerbrugge, P. M., den Boer, M. L., Kuiper, R. P., Moorman, A. V., Boer, J. M., & van Leeuwen, F. N. (2023). The Prognostic Effect of IKZF1 Deletions in ETV6::RUNX1 and High Hyperdiploid Childhood Acute Lymphoblastic Leukemia. HEMASPHERE, 7(5), e875. https://doi.org/10.1097/HS9.0000000000000875

Vancouver

Østergaard A, Enshaei A, Pieters R, Vora A, Horstmann MA, Escherich G et al. The Prognostic Effect of IKZF1 Deletions in ETV6::RUNX1 and High Hyperdiploid Childhood Acute Lymphoblastic Leukemia. HEMASPHERE. 2023 May;7(5):e875. https://doi.org/10.1097/HS9.0000000000000875

Bibtex

@article{7c71ca2c2bae4530a23495f121827489,
title = "The Prognostic Effect of IKZF1 Deletions in ETV6::RUNX1 and High Hyperdiploid Childhood Acute Lymphoblastic Leukemia",
abstract = "IKZF1 deletions are an established prognostic factor in childhood acute lymphoblastic leukemia (ALL). However, their relevance in patients with good risk genetics, namely ETV6::RUNX1 and high hyperdiploid (HeH), ALL remains unclear. We assessed the prognostic impact of IKZF1 deletions in 939 ETV6::RUNX1 and 968 HeH ALL patients by evaluating data from 16 trials from 9 study groups. Only 3% of ETV6::RUNX1 cases (n = 26) were IKZF1-deleted; this adversely affected survival combining all trials (5-year event-free survival [EFS], 79% versus 92%; P = 0.02). No relapses occurred among the 14 patients with an IKZF1 deletion treated on a minimal residual disease (MRD)-guided protocols. Nine percent of HeH cases (n = 85) had an IKZF1 deletion; this adversely affected survival in all trials (5-year EFS, 76% versus 89%; P = 0.006) and in MRD-guided protocols (73% versus 88%; P = 0.004). HeH cases with an IKZF1 deletion had significantly higher end of induction MRD values (P = 0.03). Multivariate Cox regression showed that IKZF1 deletions negatively affected survival independent of sex, age, and white blood cell count at diagnosis in HeH ALL (hazard ratio of relapse rate [95% confidence interval]: 2.48 [1.32-4.66]). There was no evidence to suggest that IKZF1 deletions affected outcome in the small number of ETV6::RUNX1 cases in MRD-guided protocols but that they are related to higher MRD values, higher relapse, and lower survival rates in HeH ALL. Future trials are needed to study whether stratifying by MRD is adequate for HeH patients or additional risk stratification is necessary.",
author = "Anna {\O}stergaard and Amir Enshaei and Rob Pieters and Ajay Vora and Horstmann, {Martin A} and Gabriele Escherich and Bertil Johansson and Mats Heyman and Kjeld Schmiegelow and Hoogerbrugge, {Peter M} and {den Boer}, {Monique L} and Kuiper, {Roland P} and Moorman, {Anthony V} and Boer, {Judith M} and {van Leeuwen}, {Frank N}",
note = "Copyright {\textcopyright} 2023 the Author(s). Published by Wolters Kluwer Health, Inc. on behalf of the European Hematology Association.",
year = "2023",
month = may,
doi = "10.1097/HS9.0000000000000875",
language = "English",
volume = "7",
pages = "e875",
journal = "HEMASPHERE",
issn = "2572-9241",
publisher = "Wolters Kluwer Health",
number = "5",

}

RIS

TY - JOUR

T1 - The Prognostic Effect of IKZF1 Deletions in ETV6::RUNX1 and High Hyperdiploid Childhood Acute Lymphoblastic Leukemia

AU - Østergaard, Anna

AU - Enshaei, Amir

AU - Pieters, Rob

AU - Vora, Ajay

AU - Horstmann, Martin A

AU - Escherich, Gabriele

AU - Johansson, Bertil

AU - Heyman, Mats

AU - Schmiegelow, Kjeld

AU - Hoogerbrugge, Peter M

AU - den Boer, Monique L

AU - Kuiper, Roland P

AU - Moorman, Anthony V

AU - Boer, Judith M

AU - van Leeuwen, Frank N

N1 - Copyright © 2023 the Author(s). Published by Wolters Kluwer Health, Inc. on behalf of the European Hematology Association.

PY - 2023/5

Y1 - 2023/5

N2 - IKZF1 deletions are an established prognostic factor in childhood acute lymphoblastic leukemia (ALL). However, their relevance in patients with good risk genetics, namely ETV6::RUNX1 and high hyperdiploid (HeH), ALL remains unclear. We assessed the prognostic impact of IKZF1 deletions in 939 ETV6::RUNX1 and 968 HeH ALL patients by evaluating data from 16 trials from 9 study groups. Only 3% of ETV6::RUNX1 cases (n = 26) were IKZF1-deleted; this adversely affected survival combining all trials (5-year event-free survival [EFS], 79% versus 92%; P = 0.02). No relapses occurred among the 14 patients with an IKZF1 deletion treated on a minimal residual disease (MRD)-guided protocols. Nine percent of HeH cases (n = 85) had an IKZF1 deletion; this adversely affected survival in all trials (5-year EFS, 76% versus 89%; P = 0.006) and in MRD-guided protocols (73% versus 88%; P = 0.004). HeH cases with an IKZF1 deletion had significantly higher end of induction MRD values (P = 0.03). Multivariate Cox regression showed that IKZF1 deletions negatively affected survival independent of sex, age, and white blood cell count at diagnosis in HeH ALL (hazard ratio of relapse rate [95% confidence interval]: 2.48 [1.32-4.66]). There was no evidence to suggest that IKZF1 deletions affected outcome in the small number of ETV6::RUNX1 cases in MRD-guided protocols but that they are related to higher MRD values, higher relapse, and lower survival rates in HeH ALL. Future trials are needed to study whether stratifying by MRD is adequate for HeH patients or additional risk stratification is necessary.

AB - IKZF1 deletions are an established prognostic factor in childhood acute lymphoblastic leukemia (ALL). However, their relevance in patients with good risk genetics, namely ETV6::RUNX1 and high hyperdiploid (HeH), ALL remains unclear. We assessed the prognostic impact of IKZF1 deletions in 939 ETV6::RUNX1 and 968 HeH ALL patients by evaluating data from 16 trials from 9 study groups. Only 3% of ETV6::RUNX1 cases (n = 26) were IKZF1-deleted; this adversely affected survival combining all trials (5-year event-free survival [EFS], 79% versus 92%; P = 0.02). No relapses occurred among the 14 patients with an IKZF1 deletion treated on a minimal residual disease (MRD)-guided protocols. Nine percent of HeH cases (n = 85) had an IKZF1 deletion; this adversely affected survival in all trials (5-year EFS, 76% versus 89%; P = 0.006) and in MRD-guided protocols (73% versus 88%; P = 0.004). HeH cases with an IKZF1 deletion had significantly higher end of induction MRD values (P = 0.03). Multivariate Cox regression showed that IKZF1 deletions negatively affected survival independent of sex, age, and white blood cell count at diagnosis in HeH ALL (hazard ratio of relapse rate [95% confidence interval]: 2.48 [1.32-4.66]). There was no evidence to suggest that IKZF1 deletions affected outcome in the small number of ETV6::RUNX1 cases in MRD-guided protocols but that they are related to higher MRD values, higher relapse, and lower survival rates in HeH ALL. Future trials are needed to study whether stratifying by MRD is adequate for HeH patients or additional risk stratification is necessary.

U2 - 10.1097/HS9.0000000000000875

DO - 10.1097/HS9.0000000000000875

M3 - SCORING: Journal article

C2 - 37153875

VL - 7

SP - e875

JO - HEMASPHERE

JF - HEMASPHERE

SN - 2572-9241

IS - 5

ER -