Zhang ZR, Dou AX, Liu Y, Zhu HB, Jia HP, Kong QH, Sun LK, Qin AQ. Hemophagocytic lymphohistiocytosis after autologous stem cell transplantation in angioimmunoblastic T-cell lymphoma: A case report. World J Clin Cases 2023; 11(17): 4072-4078 [PMID: 37388798 DOI: 10.12998/wjcc.v11.i17.4072]
Corresponding Author of This Article
Ai-Qiong Qin, MM, Doctor, Department of Cadres Medical Care/ Gerontology Geriatric, The Second Hospital of Shandong University, Cheeloo College of Medicine, Shandong University, No. 247 Beiyuan street, Jinan 250033, Shandong Province, China. qaqdax20221014@163.com
Research Domain of This Article
Oncology
Article-Type of This Article
Case Report
Open-Access Policy of This Article
This article is an open-access article which was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution Non Commercial (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/
Zhi-Rong Zhang, Yan Liu, Hong-Bo Zhu, Hai-Peng Jia, Qiu-Hong Kong, Department of Haematology, The Second Affiliated Hospital of Shandong First Medical University, Ta’an 271000, Shandong Province, China
Ai-Xia Dou, Li-Kun Sun, Department of Haematology, The Second Hospital of Shandong University, Cheeloo College of Medicine, Shandong University, Jinan 250033, Shandong Province, China
Ai-Qiong Qin, Department of Cadres Medical Care/Gerontology Geriatric, The Second Hospital of Shandong University, Cheeloo College of Medicine, Shandong University, Jinan 250033, Shandong Province, China
Author contributions: Qin AQ and Dou AX performed manuscript writing and editing; Zhang ZR, Liu Y, Zhu HB, and Jia HP performed autologous stem cell transplantation and clinical treatment; Kong QH and Sun LK performed peripheral blood stem cell collection; Dou AX provided the research funds.
Supported bythe Jinan Clinical Medical Science and Technology Innovation Plan, No. 202019141; and Norman Bethune Foundation-Feifan Iron Supplement Project, No. ffbt-C-2022-010.
Informed consent statement: Informed written consent was obtained from the patient for the publication of this report and all 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), 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: https://creativecommons.org/Licenses/by-nc/4.0/
Corresponding author: Ai-Qiong Qin, MM, Doctor, Department of Cadres Medical Care/ Gerontology Geriatric, The Second Hospital of Shandong University, Cheeloo College of Medicine, Shandong University, No. 247 Beiyuan street, Jinan 250033, Shandong Province, China. qaqdax20221014@163.com
Received: February 6, 2023 Peer-review started: February 6, 2023 First decision: April 26, 2023 Revised: May 9, 2023 Accepted: May 16, 2023 Article in press: May 16, 2023 Published online: June 16, 2023 Processing time: 126 Days and 1.7 Hours
Abstract
BACKGROUND
Angioimmunoblastic T-cell lymphoma (AITL), a unique subtype of peripheral T-cell lymphoma, has relatively poor outcomes. High-dose chemotherapy with autologous stem cell transplantation (ASCT) can achieve complete remission and improve outcomes. Unfortunately, subsequent T-cell lymphoma-triggered hemophagocytic lymphohistiocytosis (HLH) has a worse prognosis than B-cell lymphoma-triggered HLH.
CASE SUMMARY
We here report a 50-year-old woman with AITL who achieved a favorable outcome after developing HLH 2 mo after receiving high-dose chemotherapy/ ASCT. The patient was initially admitted to our hospital because of multiple enlarged lymph nodes. The final pathologic diagnosis, made on biopsy of a left axillary lymph node was AITL (Stage IV, Group A). Four cycles of the following chemotherapy regimen were administered: Cyclophosphamide 1.3 g, doxorubicin 86 mg, and vincristine 2 mg on day 1; prednisone 100 mg on days 1-5; and lenalidomide 25 mg on days 1-14. The interval between each cycle was 21 d. The patient received a conditioning regimen (busulfan, cyclophosphamide, and etoposide) followed by peripheral blood stem cell infusion. Unfortunately, she developed sustained fever and a low platelet count 17 d after ACST, leading to a diagnosis of HLH after ASCT. During treatment, she experienced thrombocytopenia and Pneumocystis carinii pneumonia. The patient was successfully treated with etoposide and glucocorticoids.
CONCLUSION
It is possible that development of HLH is related to immune reconstitution after ASCT.
Core Tip: Angioimmunoblastic T cell lymphoma (AITL), a subtype of mature peripheral T cell lymphoma, is characterized by intense inflammatory and immune reactions. Although high-dose chemotherapy with autologous stem cell transplantation (ASCT) is currently considered the optimal treatment option for AITL, subsequent development of hemophagocytic lymphohistiocytosis (HLH), a rare, life-threatening immunological syndrome, may still result in poor outcomes. In the present case, we speculate that HLH was likely attributable to immune reconstitution after ASCT. Successful immune reconstitution after ASCT is essential to improving overall survival and preventing opportunistic infections in patients with AITL.