Therapy-related myeloid neoplasms in children and adolescents

Hee Won Cho; Young Bae Choi; Eun Sang Yi; Ji Won Lee; Ki Woong Sung; Hong Hoe Koo; Keon Hee Yoo

doi:10.5045/br.2016.51.4.242

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Original Article

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Blood Res 2016; 51(4):

Published online December 23, 2016

https://doi.org/10.5045/br.2016.51.4.242

Therapy-related myeloid neoplasms in children and adolescents

Hee Won Cho¹, Young Bae Choi², Eun Sang Yi¹, Ji Won Lee¹, Ki Woong Sung¹, Hong Hoe Koo¹, and Keon Hee Yoo^1,3*

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¹Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea.

²Department of Pediatrics, Chung-Ang University Hospital, Seoul, Korea.

³Department of Medical Device Management and Research, SAIHST, Sungkyunkwan University, Seoul, Korea.

Correspondence to : Keon Hee Yoo, M.D., Ph.D. Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine, 81 Irwon-ro, Gangnam-gu, Seoul 06351, Korea. hema2170@skku.edu

Received: May 17, 2016; Revised: August 18, 2016; Accepted: September 6, 2016

This is an Open Access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (http://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract

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Abstract

Background

This retrospective study aimed to characterize and analyze the outcome of therapy-related myeloid neoplasms (t-MNs) in children and adolescents.

Methods

The medical records of 16 patients under 21 years of age at the time of t-MN diagnosis were reviewed.

Results

The median patient age was 11.5 years (range, 1.6–20.4 yr). Twelve patients had therapy-related acute myeloid leukemia, 3 patients had myelodysplastic syndrome, and 1 patient had chronic myelomonocytic leukemia. The median latency period was 29 months (range, 11–68 mo). Fourteen patients had cytogenetic aberrations, 8 of whom had an 11q23 abnormality. Of the 13 patients treated with curative intent, 12 patients received myeloid-type induction therapy that led to complete remission (CR) in 8 patients. Nine patients underwent allogeneic transplantation; 4 patients did not undergo transplantation due to chemotherapy-related toxic death (N=3) or parental refusal (N=1). The 5-year overall survival and event-free survival of the 13 patients treated with a curative intent were 46.2% and 30.8%, respectively. For the 9 patients who underwent allogeneic transplantation, the 5-year event-free survival was 66.7%.

Conclusion

A significant proportion of young patients with t-MNs can experience long-term survival, and allogeneic transplantation plays a key role for attaining cure in these patients.

Keywords Therapy-related myeloid neoplasms, Pediatric population, Allogeneic hematopoietic stem cell transplantation

Article

Original Article

Blood Res 2016; 51(4): 242-248

Published online December 23, 2016 https://doi.org/10.5045/br.2016.51.4.242

Therapy-related myeloid neoplasms in children and adolescents

Hee Won Cho¹, Young Bae Choi², Eun Sang Yi¹, Ji Won Lee¹, Ki Woong Sung¹, Hong Hoe Koo¹, and Keon Hee Yoo^1,3*

¹Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine, Seoul, Korea.

²Department of Pediatrics, Chung-Ang University Hospital, Seoul, Korea.

³Department of Medical Device Management and Research, SAIHST, Sungkyunkwan University, Seoul, Korea.

Correspondence to: Keon Hee Yoo, M.D., Ph.D. Department of Pediatrics, Samsung Medical Center, Sungkyunkwan University School of Medicine, 81 Irwon-ro, Gangnam-gu, Seoul 06351, Korea. hema2170@skku.edu

Received: May 17, 2016; Revised: August 18, 2016; Accepted: September 6, 2016

Abstract

Background

This retrospective study aimed to characterize and analyze the outcome of therapy-related myeloid neoplasms (t-MNs) in children and adolescents.

Methods

The medical records of 16 patients under 21 years of age at the time of t-MN diagnosis were reviewed.

Results

Conclusion

A significant proportion of young patients with t-MNs can experience long-term survival, and allogeneic transplantation plays a key role for attaining cure in these patients.

Keywords: Therapy-related myeloid neoplasms, Pediatric population, Allogeneic hematopoietic stem cell transplantation

Abstract

Fig 1.

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Figure 1.

Overall survival (OS) and event-free survival (EFS) for 13 patients who were treated with a curative intent. The 5-year OS was 46.2%, and the 5-year EFS was 30.8%.

Blood Research 2016; 51: 242-248https://doi.org/10.5045/br.2016.51.4.242

Fig 2.

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Figure 2.

Overall survival (OS) and event-free survival (EFS) for 9 patients who underwent allogeneic hematopoietic stem cell transplantation. The 5-year OS was 66.7% and the 5-year EFS was 66.7%.

Blood Research 2016; 51: 242-248https://doi.org/10.5045/br.2016.51.4.242

Fig 3.

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Figure 3.

The event-free survival (EFS) after allogeneic hematopoietic stem cell transplantation in terms of disease status at the time of transplantation. The 5-year EFS rate was not significantly different between CR1/CR2 (N=5) and persistent disease status (N=4) (P=0.478).

Abbreviations: CR1, complete remission 1; CR2, complete remission 2.

Blood Research 2016; 51: 242-248https://doi.org/10.5045/br.2016.51.4.242

Table 1 . Characteristics of 16 patients with therapy-related myeloid neoplasms..

Abbreviations: t-MNs, therapy-related myeloid neoplasms; ALL, acute lymphoblastic leukemia; ABL, acute biphenotypic leukemia; NHL, non-Hodgkin lymphoma; JMML, juvenile myelomonocytic leukemia; AML, acute myeloid leukemia; t-AML, therapy-related acute myeloid leukemia; t-CMML, therapy-related chronic myelomonocytic leukemia..

Table 2 . Description of previous therapeutic exposures and cytogenetics in 16 patients with therapy-related myeloid neoplasms..

^a)Latency period was defined as the interval from the first diagnosis of the primary disease to the diagnosis of t-MNs..

Abbreviations: t-AML, therapy-related acute myeloid leukemia; ABL, acute biphenotypic leukemia; DOX, doxorubicin; VP16, etoposide; MLL abn., 11q23 abnormalities; t-CMML, therapy-related chronic myelomonocytic leukemia; RMS, rhabdomyosarcoma; N/A, not available; OSA, osteosarcoma; NHL, non-Hodgkin lymphoma; ALL, acute lymphoblastic leukemia; DAU, daunorubicin; t-MDS, therapy-related myelodysplastic syndrome; SS, synovial sarcoma; NHL, non-Hodgkin lymphoma; AML, acute myeloid leukemia; IDA, idarubicin; MXT, mitoxantrone; EWS, Ewing sarcoma; BT, brain tumor; JMML, juvenile myelomonocytic leukemia; RMS, rhabdomyosarcoma; HBL, hepatoblastoma..

Table 3 . Characteristics of 13 patients with therapy-related myeloid neoplasms treated with a curative intent..

Abbreviations: HSCT, hematopoietic stem cell transplantation; t-MNs, therapy-related myeloid neoplasms; t-AML, therapy-related acute myeloid leukemia; IDA, idarubicin; BH-AC, N4-behenoyl-1-beta-D-arabinofuranosylcytosine; CR, complete remission; N/A, not applicable; t-CMML, therapy-related chronic myelomonocytic leukemia; mATV, modified A-triple-V regimen; Ara-C, cytarabine; Bu, busulfan; Cy, cyclophosphamide; VP16, etoposide; MRD, matched-related donor; PBSC, peripheral blood stem cell; Flu, fludarabine; t-MDS, therapy-related myelodysplastic syndrome; TBI, total body irradiation; rATG, rabbit antithymocyte globulin; MUD, matched-unrelated donor; UCB, umbilical cord blood; Mel, melphalan..