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ORIGINAL RESEARCH article

Front. Pediatr.
Sec. Pediatric Hematology and Hematological Malignancies
Volume 13 - 2025 | doi: 10.3389/fped.2025.1530832

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

Provisionally accepted
Eri Ushida Eri Ushida 1Hidemi Toyoda Hidemi Toyoda 1*Kohso Atsushi Kohso Atsushi 1Yosuke Okumura Yosuke Okumura 1Kaori Niwa Kaori Niwa 1Takahiro Ito Takahiro Ito 1Mari Morimoto Mari Morimoto 1Ryo Hanaki Ryo Hanaki 1Keishiro Amano Keishiro Amano 1Shotaro Iwamoto Shotaro Iwamoto 1Takao Deguchi Takao Deguchi 2Hiroki Hori Hiroki Hori 3Masahiro Hirayama Masahiro Hirayama 1
  • 1 Mie University, Tsu, Japan
  • 2 National Center for Child Health and Development (NCCHD), Tokyo, Japan
  • 3 Suzuka University of Medical Science, Suzuka, Mie, Japan

The final, formatted version of the article will be published soon.

    Background: Acute 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. Procedure: We 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. Results: Ten 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). Conclusions: As 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.

    Keywords: Acute Lymphoblastic Leukemia, Lymphoblastic lymphoma, Secondary neoplasm, cranial radiotherapy, total body irradiation Abbreviations ALL acute lymphoblastic leukemia allo-HCT allogeneic hematopoietic stem-cell transplantation CNS central nervous system CRT cranial radiotherapy LBL lymphoblastic lymphoma

    Received: 19 Nov 2024; Accepted: 08 Jan 2025.

    Copyright: © 2025 Ushida, Toyoda, Atsushi, Okumura, Niwa, Ito, Morimoto, Hanaki, Amano, Iwamoto, Deguchi, Hori and Hirayama. This is an open-access article distributed under the terms of the Creative Commons Attribution License (CC BY). The use, distribution or reproduction in other forums is permitted, provided the original author(s) or licensor are credited and that the original publication in this journal is cited, in accordance with accepted academic practice. No use, distribution or reproduction is permitted which does not comply with these terms.

    * Correspondence: Hidemi Toyoda, Mie University, Tsu, Japan

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