Secondary neoplasms in survivors of pediatric acute lymphoblastic leukemia and lymphoblastic lymphoma: a single-center, retrospective study

BackgroundAcute lymphoblastic leukemia (ALL)-based therapeutic regimens have markedly improved the survival of children with ALL and lymphoblastic lymphoma (LBL). However, survivors are at risk of secondary neoplasms. Few studies on such secondary neoplasms have been conducted outside of Europe and...

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Main Authors: Eri Ushida, Hidemi Toyoda, Atsushi Kohso, Yosuke Okumura, Kaori Niwa, Takahiro Ito, Mari Morimoto, Ryo Hanaki, Keishiro Amano, Shotaro Iwamoto, Takao Deguchi, Hiroki Hori, Masahiro Hirayama
Format: Article
Language:English
Published: Frontiers Media S.A. 2025-01-01
Series:Frontiers in Pediatrics
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Online Access:https://www.frontiersin.org/articles/10.3389/fped.2025.1530832/full
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author Eri Ushida
Hidemi Toyoda
Atsushi Kohso
Yosuke Okumura
Kaori Niwa
Takahiro Ito
Mari Morimoto
Ryo Hanaki
Keishiro Amano
Shotaro Iwamoto
Takao Deguchi
Hiroki Hori
Masahiro Hirayama
author_facet Eri Ushida
Hidemi Toyoda
Atsushi Kohso
Yosuke Okumura
Kaori Niwa
Takahiro Ito
Mari Morimoto
Ryo Hanaki
Keishiro Amano
Shotaro Iwamoto
Takao Deguchi
Hiroki Hori
Masahiro Hirayama
author_sort Eri Ushida
collection DOAJ
description BackgroundAcute lymphoblastic leukemia (ALL)-based therapeutic regimens have markedly improved the survival of children with ALL and lymphoblastic lymphoma (LBL). However, survivors are at risk of secondary neoplasms. Few studies on such secondary neoplasms have been conducted outside of Europe and the United States. The aim of this study was to evaluate the incidence of, risk factors for, and outcomes of secondary neoplasms in long-term survivors of ALL and LBL at a tertiary pediatric oncology center in Mie prefecture, Japan.ProcedureWe retrospectively reviewed 188 patients with ALL and LBL who were treated with an ALL-based therapeutic regimen at Mie University Hospital from January 1, 1977 to December 31, 2022 and followed up.ResultsTen patients developed secondary neoplasms, with 10-year and 20-year cumulative incidences of 2.9% [standard error (SE) = 1.5%] and 5.5% (SE = 2.3%), respectively. The median interval between the primary-cancer diagnosis and secondary-neoplasm diagnosis was 18.5 years (range: 7.8–41.7 years). All 10 secondary neoplasms were central nervous system (CNS) tumors (6 meningiomas and 4 high-grade gliomas). Radiotherapy (p = 0.007) and CNS involvement in the primary cancer (p < 0.001) increased the risk of secondary neoplasms among long-term survivors. Gliomas occurred significantly earlier than meningiomas (p = 0.047), and three patients died of secondary neoplasms (all gliomas).ConclusionsAs secondary gliomas occurred earlier than meningiomas and are associated with poor outcomes, physicians should take great pains to minimize their risk to improve long-term survival and quality of life.
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spelling doaj-art-ab44e8eee0d14cfda68c983aeed8754a2025-01-28T06:41:03ZengFrontiers Media S.A.Frontiers in Pediatrics2296-23602025-01-011310.3389/fped.2025.15308321530832Secondary neoplasms in survivors of pediatric acute lymphoblastic leukemia and lymphoblastic lymphoma: a single-center, retrospective studyEri Ushida0Hidemi Toyoda1Atsushi Kohso2Yosuke Okumura3Kaori Niwa4Takahiro Ito5Mari Morimoto6Ryo Hanaki7Keishiro Amano8Shotaro Iwamoto9Takao Deguchi10Hiroki Hori11Masahiro Hirayama12Department of Pediatrics, Mie University Graduate School of Medicine, Tsu, JapanDepartment of Pediatrics, Mie University Graduate School of Medicine, Tsu, JapanDepartment of Pediatrics, Mie University Graduate School of Medicine, Tsu, JapanDepartment of Pediatrics, Mie University Graduate School of Medicine, Tsu, JapanDepartment of Pediatrics, Mie University Graduate School of Medicine, Tsu, JapanDepartment of Pediatrics, Mie University Graduate School of Medicine, Tsu, JapanDepartment of Pediatrics, Mie University Graduate School of Medicine, Tsu, JapanDepartment of Pediatrics, Mie University Graduate School of Medicine, Tsu, JapanDepartment of Pediatrics, Mie University Graduate School of Medicine, Tsu, JapanDepartment of Pediatrics, Mie University Graduate School of Medicine, Tsu, JapanDivision of Cancer Immunodiagnostics, Children’s Cancer Center, National Center for Child Health and Development, Tokyo, JapanDepartment of Medical Technology Course, Suzuka University of Medical Science, Suzuka, JapanDepartment of Pediatrics, Mie University Graduate School of Medicine, Tsu, JapanBackgroundAcute lymphoblastic leukemia (ALL)-based therapeutic regimens have markedly improved the survival of children with ALL and lymphoblastic lymphoma (LBL). However, survivors are at risk of secondary neoplasms. Few studies on such secondary neoplasms have been conducted outside of Europe and the United States. The aim of this study was to evaluate the incidence of, risk factors for, and outcomes of secondary neoplasms in long-term survivors of ALL and LBL at a tertiary pediatric oncology center in Mie prefecture, Japan.ProcedureWe retrospectively reviewed 188 patients with ALL and LBL who were treated with an ALL-based therapeutic regimen at Mie University Hospital from January 1, 1977 to December 31, 2022 and followed up.ResultsTen patients developed secondary neoplasms, with 10-year and 20-year cumulative incidences of 2.9% [standard error (SE) = 1.5%] and 5.5% (SE = 2.3%), respectively. The median interval between the primary-cancer diagnosis and secondary-neoplasm diagnosis was 18.5 years (range: 7.8–41.7 years). All 10 secondary neoplasms were central nervous system (CNS) tumors (6 meningiomas and 4 high-grade gliomas). Radiotherapy (p = 0.007) and CNS involvement in the primary cancer (p < 0.001) increased the risk of secondary neoplasms among long-term survivors. Gliomas occurred significantly earlier than meningiomas (p = 0.047), and three patients died of secondary neoplasms (all gliomas).ConclusionsAs secondary gliomas occurred earlier than meningiomas and are associated with poor outcomes, physicians should take great pains to minimize their risk to improve long-term survival and quality of life.https://www.frontiersin.org/articles/10.3389/fped.2025.1530832/fullacute lymphoblastic leukemialymphoblastic lymphomasecondary neoplasmcranial radiotherapytotal body irradiation
spellingShingle Eri Ushida
Hidemi Toyoda
Atsushi Kohso
Yosuke Okumura
Kaori Niwa
Takahiro Ito
Mari Morimoto
Ryo Hanaki
Keishiro Amano
Shotaro Iwamoto
Takao Deguchi
Hiroki Hori
Masahiro Hirayama
Secondary neoplasms in survivors of pediatric acute lymphoblastic leukemia and lymphoblastic lymphoma: a single-center, retrospective study
Frontiers in Pediatrics
acute lymphoblastic leukemia
lymphoblastic lymphoma
secondary neoplasm
cranial radiotherapy
total body irradiation
title Secondary neoplasms in survivors of pediatric acute lymphoblastic leukemia and lymphoblastic lymphoma: a single-center, retrospective study
title_full Secondary neoplasms in survivors of pediatric acute lymphoblastic leukemia and lymphoblastic lymphoma: a single-center, retrospective study
title_fullStr Secondary neoplasms in survivors of pediatric acute lymphoblastic leukemia and lymphoblastic lymphoma: a single-center, retrospective study
title_full_unstemmed Secondary neoplasms in survivors of pediatric acute lymphoblastic leukemia and lymphoblastic lymphoma: a single-center, retrospective study
title_short Secondary neoplasms in survivors of pediatric acute lymphoblastic leukemia and lymphoblastic lymphoma: a single-center, retrospective study
title_sort secondary neoplasms in survivors of pediatric acute lymphoblastic leukemia and lymphoblastic lymphoma a single center retrospective study
topic acute lymphoblastic leukemia
lymphoblastic lymphoma
secondary neoplasm
cranial radiotherapy
total body irradiation
url https://www.frontiersin.org/articles/10.3389/fped.2025.1530832/full
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