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复发或难治性弥漫性大B细胞淋巴瘤患者接受嵌合抗原受体T细胞治疗后的颈部局部细胞因子释放综合征

Cervical Local Cytokine Release Syndrome Following Chimeric Antigen Receptor T-cell Therapy in Patients With Relapsed or Refractory Diffuse Large B-cell Lymphoma.

作者信息

Inoue Yu, Fujino Takahiro, Chinen Shotaro, Niiyama-Uchibori Yui, Ide Daisuke, Kawata Moe, Hashimoto Keiko, Takimoto-Shimomura Tomoko, Nakayama Ai, Tsukamoto Taku, Mizutani Shinsuke, Shimura Yuji, Hirano Shigeru, Kuroda Junya

机构信息

Division of Hematology and Oncology, Department of Medicine, Kyoto Prefectural University of Medicine, Kyoto, JPN.

Department of Otolaryngology-Head and Neck Surgery, Kyoto Prefectural University of Medicine, Kyoto, JPN.

出版信息

Cureus. 2023 May 11;15(5):e38905. doi: 10.7759/cureus.38905. eCollection 2023 May.


DOI:10.7759/cureus.38905
PMID:37303362
原文链接:https://pmc.ncbi.nlm.nih.gov/articles/PMC10257532/
Abstract

The use of chimeric antigen receptor T-cell (CAR-T) therapy for hematologic malignancies is rapidly increasing, and appropriately managing adverse events (AEs) is crucial. Cytokine release syndrome (CRS) is a common AE of CAR-T therapy, characterized by systemic symptoms such as fever and respire-circulatory failure. We present two cases with relapsed or refractory diffuse large B-cell lymphoma (DLBCL) accompanied by a rare complication of cervical local CRS as an acute inflammatory reaction at a specific site after CAR-T infusion. Case 1: A 60-year-old gentleman with diffuse large B cell lymphoma (DLBCL) developed grade 1 CRS on day one that required three doses of tocilizumab. Then he developed remarkable cervical edema as local CRS on day five. His local CRS spontaneously improved from day seven without additional therapy. Case 2: A 70-year-old gentleman with DLBCL developed grade 1 CRS on day two that required three doses of tocilizumab. Then he developed remarkable cervical edema and muffled voice as local CRS on day three. He received dexamethasone because of concerns about airway obstruction, and his local CRS improved immediately after dexamethasone administration. Before Tisa-Cel infusion, neither patients had a lymphoma lesion in their necks. To summarize, local CRS may occur at the site without lymphoma involvement after CAR-T therapy. An appropriate diagnosis and careful observation are required to determine the need for additional treatment.

摘要

嵌合抗原受体T细胞(CAR-T)疗法在血液系统恶性肿瘤治疗中的应用正在迅速增加,妥善处理不良事件(AE)至关重要。细胞因子释放综合征(CRS)是CAR-T疗法常见的不良事件,其特征为发热和呼吸循环衰竭等全身症状。我们报告两例复发或难治性弥漫性大B细胞淋巴瘤(DLBCL)患者,在接受CAR-T输注后,出现罕见的颈部局部CRS并发症,表现为特定部位的急性炎症反应。病例1:一名60岁患有弥漫性大B细胞淋巴瘤(DLBCL)的男性患者在第1天出现1级CRS,需要使用三剂托珠单抗。然后在第5天,他出现明显的颈部水肿,为局部CRS。从第7天起,其局部CRS未经额外治疗自行改善。病例2:一名70岁患有DLBCL的男性患者在第2天出现1级CRS,需要使用三剂托珠单抗。然后在第3天,他出现明显的颈部水肿和声音嘶哑,为局部CRS。由于担心气道阻塞,他接受了地塞米松治疗,使用地塞米松后其局部CRS立即改善。在输注Tisa-Cel之前,两名患者颈部均无淋巴瘤病灶。总之,CAR-T治疗后,局部CRS可能发生在无淋巴瘤累及的部位。需要进行适当的诊断和仔细观察,以确定是否需要额外治疗。

https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f08a/10257532/97b43b47a9d5/cureus-0015-00000038905-i04.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f08a/10257532/ef7ac621ed40/cureus-0015-00000038905-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f08a/10257532/dc5d54e32b9b/cureus-0015-00000038905-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f08a/10257532/f1526f01f1a1/cureus-0015-00000038905-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f08a/10257532/97b43b47a9d5/cureus-0015-00000038905-i04.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f08a/10257532/ef7ac621ed40/cureus-0015-00000038905-i01.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f08a/10257532/dc5d54e32b9b/cureus-0015-00000038905-i02.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f08a/10257532/f1526f01f1a1/cureus-0015-00000038905-i03.jpg
https://cdn.ncbi.nlm.nih.gov/pmc/blobs/f08a/10257532/97b43b47a9d5/cureus-0015-00000038905-i04.jpg

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引用本文的文献

[1]
Severe cases of local cytokine release syndrome (CRS); craniocervical edema soon after chimeric antigen T-cell (CAR-T) therapy.

Oxf Med Case Reports. 2025-1-18

[2]
Epcoritamab-Induced fatal pleural effusion in diffuse large B-Cell lymphoma: a case report and literature review.

Ann Hematol. 2025-3

[3]
Eleven cases of laryngeal edema after tisagenlecleucel infusion: a 3-year single center retrospective study of CD19-directed chimeric antigen receptor T-cell therapy for relapsed and refractory B-cell lymphomas.

Haematologica. 2025-3-1

[4]
Salivary gland swelling as a characteristic manifestation of local cytokine release syndrome after anti-CD19 chimeric antigen receptor T cell therapy: A case series.

J Clin Exp Hematop. 2024

[5]
CT Demonstration of Local Cytokine-Release Syndrome Involving the Head and Neck Following Chimeric Antigen Receptor T Cell Infusion Therapy.

Korean J Radiol. 2024-4

本文引用的文献

[1]
The pathogenesis, diagnosis, prevention, and treatment of CAR-T cell therapy-related adverse reactions.

Front Pharmacol. 2022-10-14

[2]
Cervical Edema Extending to the Larynx as Local Cytokine Release Syndrome Following Chimeric Antigen Receptor T-Cell Therapy in a Boy with Refractory Acute Lymphoblastic Leukemia.

Case Rep Oncol. 2022-3-15

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Lancet Oncol. 2021-10

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Case Report: Local Cytokine Release Syndrome in an Acute Lymphoblastic Leukemia Patient After Treatment With Chimeric Antigen Receptor T-Cell Therapy: A Possible Model, Literature Review and Perspective.

Front Immunol. 2021

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Biol Blood Marrow Transplant. 2018-12-23

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