Case Report
Copyright ©The Author(s) 2022. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Clin Cases. Oct 6, 2022; 10(28): 10339-10345
Published online Oct 6, 2022. doi: 10.12998/wjcc.v10.i28.10339
Sudden extramedullary and extranodal Philadelphia-positive anaplastic large-cell lymphoma transformation during imatinib treatment for CML: A case report
Qiong Wu, Yong Kang, Jing Xu, Wen-Can Ye, Zhen-Jiang Li, Wen-Feng He, Yuan Song, Qing-Ming Wang, Ai-Ping Tang, Ting Zhou
Qiong Wu, Department of Clinical Laboratory, The Second Affiliated Hospital of Nanchang University, Nanchang 330006, Jiangxi Province, China
Yong Kang, Jing Xu, Wen-Can Ye, Zhen-Jiang Li, Yuan Song, Qing-Ming Wang, Ai-Ping Tang, Department of Hematology, The Second Affiliated Hospital of Nanchang University, Nanchang 330006, Jiangxi Province, China
Yong Kang, Department of Hematology, Pingxiang People's hospital, Pingxiang 337000, Jiangxi Province, China
Wen-Can Ye, Department of Hematology, The First Affiliated Hospital of Gannan Medical University, Ganzhou 341001, Jiangxi Province, China
Wen-Feng He, Key laboratory of Molecular Medicine of Jiangxi, The Second Affiliated Hospital of Nanchang University, Nanchang 330006, Jiangxi Province, China
Ting Zhou, Department of Pathology, The Second Affiliated Hospital of Nanchang University, Nanchang 330006, Jiangxi Province, China
Author contributions: Wu Q and Kang Y participated in the design of this study and drafted this manuscript; Xu J and Ye WC performed literature research and data acquisition; He WF, Song Y, Zhou T, and Tang AP provided help for the methodology used; Li ZZ and Wang QM reviewed and edited this manuscript; all authors issued final approval for the version to be submitted.
Supported by the Jiangxi “5511” Science and Technology Innovation Talent Project, No. 20171BCB18003.
Informed consent statement: Informed written consent was obtained from the patients for the publication of this report and any accompanying images.
Conflict-of-interest statement: The authors declare that they have no conflict of interest to disclose.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), according to which the manuscript was prepared and revised.
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: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Zhen-Jiang Li, PhD, Chief Doctor, Department of Hematology, The Second Affiliated Hospital of Nanchang University, No. 1 Minde Road, Nanchang 330006, Jiangxi Province, China. lzjdgh@163.com
Received: June 20, 2022
Peer-review started: June 20, 2022
First decision: July 14, 2022
Revised: July 18, 2022
Accepted: August 24, 2022
Article in press: August 24, 2022
Published online: October 6, 2022
Processing time: 98 Days and 22.5 Hours
Abstract
BACKGROUND

Chronic myeloid leukemia (CML) is a malignant hematologic malignancy that can progress to blast phase with a myeloid or lymphoid phenotype. Some patients with CML can also progress to blast crisis phase; however, the transformation of CML into Philadelphia-positive lymphoma is extremely rare.

CASE SUMMARY

We present a patient with CML who experienced a sudden transformation to anaplastic large-cell lymphoma (ALCL) after 7 mo of treatment with imatinib, during which she had achieved partial cytogenetic response as well as early molecular response. The patient noticed a mass in her left shoulder, the biopsy data of which were consistent with ALCL; moreover, her lymphoma cells exhibited BCR-ABL gene fusion. The patient was diagnosed with Philadelphia-positive ALCL that progressed from CML, and was thus treated with the second generation tyrosine kinase inhibitor nilotinib. Six months later, the mass had totally disappeared and the BCR-ABL fusion gene was undetectable in the peripheral blood. To our knowledge, this is the first patient known to have developed Philadelphia-positive ALCL transformed from CML.

CONCLUSION

Unexplained lymphadenopathy or an extramedullary mass in a patient with CML may warrant a biopsy and testing for BCR-ABL fusion.

Keywords: Chronic myeloid leukemia; BCR-ABL fusion gene; Imatinib mesylate; Anaplastic large-cell lymphoma; Philadelphia-positive; Nilotinib; Case report

Core Tip: We describe a patient with chronic myeloid leukemia (CML) whose disease underwent a sudden transformation to anaplastic large-cell lymphoma (ALCL) following partial cytogenetic remission and early molecular response with 7 months of imatinib treatment. The patient developed a mass and felt pain in her left shoulders; a biopsy of the mass revealed ALCL, and BCR/ABL fusion was detected via fluorescence in situ hybridization. The patient was then treated with nilotinib and achieved complete cytogenetic remission and undetectable molecular residual disease within 6 mo. To the best of our knowledge, our patient is the first reported to have Philadelphia-positive ALCL transformed from CML.