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Laryngeal edema as a symptom of local cytokine release syndrome after BCMA-targeting CAR-T therapy for relapsed and refractory multiple myeloma

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Abstract

Cytokine release syndrome (CRS) can be a major side effect of chimeric antigen receptor T-cell (CAR-T) therapy, and may occasionally become life-threatening in patients with factors such as high tumor burden or poor performance status. Among the many CRS events observed in B-cell maturation antigen (BCMA)-targeting CAR-T therapy, local symptoms (also called local CRS) are poorly understood due to their low frequency. Here, we present the case of a 54-year-old woman with refractory multiple myeloma exhibiting laryngeal edema as a local CRS. Before CAR-T therapy, she was diagnosed with progressive disease indicated by a left thyroid mass. After local irradiation, she received the BCMA-targeting CAR-T agent idecabtagene vicleucel (ide-cel). On day 2, the patient developed CRS, which resolved on treatment with tocilizumab. However, on day 4, laryngeal edema worsened, and was judged to be a local CRS. Intravenous dexamethasone rapidly reduced this edema. In conclusion, laryngeal edema rarely occurs as a local CRS, and to the best of our knowledge, has never been reported after ide-cel infusion. Dexamethasone was effective for reducing the local reaction that persisted after treatment of systemic symptoms with tocilizumab.

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Correspondence to Masaki Ri.

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Conflict of interest

Masaki Ri reports honoraria from Jansesen Pharmaceutical K.K. and research funds from Bristol-Myers Squibb K.K., Daichi Sankyo Co., Ltd., Celgene K.K., and scholarship endowments from Chugai Pharmaceutical Co., Ltd., Takeda Pharmaceutical Co., Ltd., Ono Pharma USA, Inc., Kyowa Kirin Co., Ltd. Shigeru Kusumoto reports honoraria from Jansesen Pharmaceutical K.K., Chugai Pharmaceutical Co., Ltd., Ono Pharma USA, Inc., and research funds from Daichi Sankyo Co., Ltd., and scholarship endowments from Chugai Pharmaceutical Co., Ltd. Shinsuke Iida reports honoraria from Bristol-Myers Squibb K.K., Janssen Pharmaceutical K.K., Sanofi Co., Ltd., Takeda Pharmaceutical Co., Ltd., Ono Pharma USA, Inc., Celgene K.K., and research funds from Bristol-Myers Squibb K.K., Janssen Pharmaceutical K.K., Sanofi Co., Ltd., Takeda Pharmaceutical Co. Ltd., Ono Pharma USA, Inc., Daichi Sankyo Co., Ltd., Abbvie GK, Novartis Pharma K.K., Otsuka Pharmaceutical K.K., Amgen Inc., Chugai Pharmaceutical Co., Ltd., Celgene K.K., Glaxo SmithKlein plc., Alexion Pharmaceuticals, Inc., Pfizer Inc., Shionogi Co., Ltd., and scholarship endowments from Chugai Pharmaceutical Co., Ltd. The other authors have nothing to report.

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Nakanishi, Y., Marumo, Y., Ri, M. et al. Laryngeal edema as a symptom of local cytokine release syndrome after BCMA-targeting CAR-T therapy for relapsed and refractory multiple myeloma. Int J Hematol 118, 647–651 (2023). https://doi.org/10.1007/s12185-023-03633-8

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  • DOI: https://doi.org/10.1007/s12185-023-03633-8

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