Vol.:(0123456789) 1 3
International Journal of Hematology
https://doi.org/10.1007/s12185-019-02599-w
ORIGINAL ARTICLE
Discontinuation of L-asparaginase and poor response to prednisolone
are associated with poor outcome of ETV6-RUNX1-positive pediatric
B-cell precursor acute lymphoblastic leukemia
Ikuya Usami
1,2
· Toshihiko Imamura
2,3
· Yoshihiro Takahashi
4
· So‑ichi Suenobu
5
· Daiichiro Hasegawa
6
·
Yoshiko Hashii
7
· Takao Deguchi
8
· Tsukasa Hori
9
· Akira Shimada
10
· Koji Kato
11
· Eturou Ito
12
· Akiko Moriya‑Saito
2
·
Hirohide Kawasaki
13
· Hiroki Hori
8
· Keiko Yumura‑Yagi
14
· Junichi Hara
15
· Atsushi Sato
16
· Keizo Horibe
2
· Japan
Association of Childhood Leukemia Study Group (JACLS)
Received: 18 September 2018 / Revised: 28 December 2018 / Accepted: 16 January 2019
© Japanese Society of Hematology 2019
Abstract
ETV6-RUNX1-positive B precursor acute lymphoblastic leukemia (B-ALL) is a common subtype of pediatric B-ALL that
has shown excellent outcomes in contemporary clinical trials for pediatric B-ALL. Examinations of the possibility of reduc-
ing therapeutic intensity may thus be explored. This prospective study examined outcomes in 205 pediatric patients with
ETV6-RUNX1-positive B-ALL uniformly treated following the Japan Association of Childhood Leukemia Study Group
(JACLS) ALL-02 protocol. The JACLS ALL-02 protocol does not employ minimal residual disease detected by polymerase
chain reaction (PCR-MRD)-based risk stratifcation; however, 4-year event-free survival (EFS) and overall survival (OS)
were 94.4 ± 1.6 and 97.5 ± 1.1%, respectively. In particular, 92 of 205 (44.9%) patients were successfully treated with a less
intensive regimen involving only two cycles of high dose methotrexate and one course of re-induction therapy comprising
vincristine, L-asparaginase (L-asp), pirarubicin, and prednisolone. Multivariate analysis revealed that discontinuation of
L-asp and poor response to prednisolone was, respectively, associated with poor EFS (HR 6.3; 95% CI 1.3–27.0) and OS
(HR 17.5; 95% CI 2.3–130), suggesting that the majority of ETV6-RUNX1-positive B-ALL cases may be cured by a less-
intensive chemotherapy regimen if the risk stratifcation system including PCR-MRD monitoring and insufcient use of
L-asp is avoided.
Keywords Poor prednisolone response · L-asparaginase · ETV6-RUNX1 · Pediatric acute lymphoblastic leukemia
* Toshihiko Imamura
imamura@koto.kpu-m.ac.jp
1
Department of Pediatric Hematology and Oncology, Hyogo
Prefectural Amagasaki General Medical Center, Hyogo,
Japan
2
Clinical Research Center, National Hospital Organization
Nagoya Medical Center, Nagoya, Japan
3
Department of Pediatrics, Graduate School of Medical
Science, Kyoto Prefectural University of Medicine, Kyoto,
Japan
4
Department of Pediatrics, Aomori Prefectural Central
Hospital, Aomori, Japan
5
Division of General Pediatrics and Emergency Medicine,
Department of Pediatrics, Oita University, Oita, Japan
6
Department of Hematology/Oncology, Hyogo Prefectural
Children’s Hospital, Kobe, Japan
7
Department of Pediatrics, Osaka University, Osaka, Japan
8
Department of Pediatrics, Mie University, Tsu, Japan
9
Department of Pediatrics, Sapporo Medical University
of Medicine, Hokkaido, Japan
10
Department of Pediatrics, Okayama University, Okayama,
Japan
11
Department of Hematology Oncology, Children’s Medical
Center, Japanese Red Cross Nagoya First Hospital, Nagoya,
Japan
12
Department of Pediatrics, Hirosaki University, Hirosaki,
Japan
13
Department of Pediatrics, Kansai Medical University,
Hirakata, Japan
14
Yumura Clinic, Osaka, Japan
15
Department of Pediatric Hematology/Oncology, Osaka City
General Hospital, Osaka, Japan
16
Department of Hematology/Oncology, Miyagi Children’s
Hospital, Sendai, Japan