• Ryoma Shimazu, Nobuhiko Nakamura, Takayuki Goto, Yuto Kaneda, Yoshikazu Ikoma, Takuro Matsumoto, Hiroshi Nakamura, Nobuhiro Kanemura, and Masahito Shimizu.
• Department of Hematology and Infectious Disease, Gifu University Hospital, Gifu, Japan.
• Medicine (Baltimore). 2024 Sep 6; 103 (36): e39630e39630.

RationaleCytokine release syndrome (CRS) is a common adverse event of chimeric antigen receptor T (CAR-T) cell therapy. CRS is generally a systemic inflammatory reaction, but in rare cases, it can occur in specific body areas and is referred to as "local CRS (L-CRS)." A case of laryngeal edema due to L-CRS that required tracheal intubation because of the lack of response to tocilizumab (TCZ) and dexamethasone (DEX) is reported.Patient ConcernsA 67-year-old woman with relapsed transformed follicular lymphoma was treated with CAR-T cell therapy. Although she had been given TCZ and DEX for CRS, neck swelling appeared on day 4 after infusion.DiagnosesLaryngoscopy showed severe laryngeal edema, which was presumed to be due to L-CRS, since there were no other apparent triggers based on history, physical examination, and computed tomography.InterventionsTracheal intubation was performed because of the risk of upper airway obstruction. Ultimately, 4 doses of tocilizumab (8 mg/kg) and 6 doses of dexamethasone (10 mg/body) were required to improve the L-CRS.OutcomesOn day 7, laryngeal edema improved, and the patient could be extubated.LessonsThe lessons from this case are, first, that CAR-T cell therapy may induce laryngeal edema in L-CRS. Second, TCZ alone may be ineffective in cervical L-CRS. Third, TCZ, as well as DEX, may be inadequate. In such cases, we should recognize L-CRS and manage it early because it may eventually progress to laryngeal edema that requires securing the airway.Copyright © 2024 the Author(s). Published by Wolters Kluwer Health, Inc.

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