Copyright
©The Author(s) 2021. Published by Baishideng Publishing Group Inc. All rights reserved.
Diagnosis and treatment of pediatric anaplastic lymphoma kinase-positive large B-cell lymphoma: A case report
Meng Zhang, Ling Jin, Yan-Long Duan, Jing Yang, Shuang Huang, Mei Jin, Guang-Hua Zhu, Chao Gao, Yi Liu, Nan Zhang, Chun-Ju Zhou, Zi-Fen Gao, Qin-Long Zheng, Dong Chen, Yong-Hong Zhang
Meng Zhang, Ling Jin, Yan-Long Duan, Jing Yang, Shuang Huang, Mei Jin, Guang-Hua Zhu, Chao Gao, Yi Liu, Nan Zhang, Yong-Hong Zhang, Beijing Key Laboratory of Pediatric Hematology Oncology, National Discipline of Pediatrics, Ministry of Education, MOE Key Laboratory of Major Diseases in Children, Hematology Oncology Center, Beijing Children’s Hospital, Capital Medical University, National Center for Children’s Health, Beijing 100045, China
Chun-Ju Zhou, Department of Pathology, Beijing Children’s Hospital, Capital Medical University, National Center for Children’s Health, Beijing 100045, China
Zi-Fen Gao, Department of Pathology, Peking University Third Hospital, Beijing 100191, China
Qin-Long Zheng, Laboratory of Molecular Diagnostics, Department of Pathology and Laboratory Medicine, Beijing Boren Hospital, Beijing 100070, China
Dong Chen, Division of Hematopathology, Mayo Clinic, Rochester, MN 55905, United States
Author contributions: Zhang YH designed the study; Zhou CJ, Gao ZF, Zheng QL, Gao C, Liu Y, and Chen D performed the assays; Zhang M, Jin L, Duan YL, Yang J, Huang S, Jin M, Zhu GH, and Zhang N collected the data and samples; Zhang M wrote the manuscript; Zhang YH reviewed the final manuscript and takes primary responsibility for the article; All authors read and approved the final manuscript.
Supported by The Special Fund of the Pediatric Medical Coordinated Development Center of Beijing Hospitals Authority, No. XTZD20180204.
Informed consent statement: Informed written consent was obtained from the patient and his parents for publication of this report and the accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflicts of interest.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See:
http://creativecommons.org/Licenses/by-nc/4.0/ Corresponding author: Yong-Hong Zhang, MD, Doctor, Beijing Key Laboratory of Pediatric Hematology Oncology, National Discipline of Pediatrics, Ministry of Education, MOE Key Laboratory of Major Diseases in Children, Hematology Oncology Center, Beijing Children’s Hospital, Capital Medical University, National Center for Children’s Health, No. 56 Nanlishi Road, Beijing 100045, China.
yhzhang58@hotmail.com
Received: December 31, 2020
Peer-review started: December 31, 2020
First decision: February 25, 2021
Revised: March 15, 2021
Accepted: March 29, 2021
Article in press: March 29, 2021
Published online: June 16, 2021
Processing time: 145 Days and 17.6 Hours
BACKGROUND
Anaplastic lymphoma kinase-positive (ALK+) large B-cell lymphoma (LBCL) is a rare type of lymphoma with high invasiveness and rapid progression. It occurs in all age groups, but is extremely rare in children. The lesions mainly involve the lymph nodes and may present with extra-nodal involvement. Response to conventional chemotherapies and local radiotherapy is poor, with a 5-year overall survival of less than 40%. Recently, the use of ALK inhibitors for the treatment of this disease has been reported.
CASE SUMMARY
We present a case of a 12-year-old boy diagnosed with ALK+LBCL. The patient had a 2-mo medical history of a calvarial mass, extensive systemic involvement, and positive bone marrow clathrin heavy chain (CLTC)-ALK fusion gene. Complete remission 1 (CR1) was achieved using the modified LMB89 Group C regimen followed by autologous stem cell transplantation. The patient relapsed 3 mo later. He then achieved CR2 with three short courses of chemotherapy (COP, reduced-dose ICE, low-dose Ara-c+VP16) and continuous alectinib targeted therapy. Afterward, allogeneic hematopoietic stem cell transplantation (allo-HSCT) was performed. At 16 mo after the allo-HSCT, the patient was still in CR2.
CONCLUSION
The modified LMB89 Group C regimen and ALK inhibitors are effective. Allo-HSCT should be performed after remission.
Core Tip: Anaplastic lymphoma kinase-positive (ALK+) large B-cell lymphoma (LBCL) is a rare subtype of diffuse LBCL and is particularly rare in pediatric non-Hodgkin lymphoma patients. The modified LMB89 Group C regimen is effective for ALK+LBCL. Since bone marrow failure occurs after relapse, the patient cannot tolerate intensive chemotherapy; thus, ALK inhibitors combined with low-dose chemotherapy could be considered, and there is still hope for complete remission 2. It is advised that allogeneic hematopoietic stem cell transplantation be performed as soon as possible after remission.
