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Management of a critical airway emergency caused by large oral haematoma secondary to severe mucositis

Caris Coyle, Heather Storey, Ismail Elbeshlawi and 3 more

EJC Paediatric Oncology · 2025

doi:10.1016/j.ejcped.2025.100250 (opens in a new tab)

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Abstract

Case report: A boy, 12 years old with refractory acute lymphocytic leukemia, underwent CAR-T cell infusion per institutional protocol.Initially asymptomatic, he developed fever, worsening renal function, elevated liver enzymes, and norepinephrine-dependent hypotensive shock 24 hours post-infusion.Based on clinical and laboratory findings, cytokine release syndrome (CRS) was diagnosed, and he received two doses of tocilizumab.On the second day, the patient developed mild mental confusion (ICANS 1), prompting dexamethasone therapy.Despite treatment, renal dysfunction progressed, leading to anasarca, intubation, and continuous venovenous hemodialysis.After catheter placement, the patient presented mydriatic pupils, unresponsive to stimuli, and was reclassified as ICANS 4. Anakinra was initiated, and imaging ruled out other abnormalities.The patient progressively improved, was weaned off norepinephrine and extubated before five days, and discharged without neurological deficits.CAR-T therapy represents a promising treatment for refractory hematologic malignancies; however, ICANS remains a frequent and potentially fatal complication, affecting up to 60% of CD19 CAR-T recipients.The pathophysiology involves inflammatory cytokines, blood-brain barrier disruption, and cerebrospinal fluid cytokine elevation, potentially leading to cerebral edema.Diagnosis is challenging, particularly in critically ill patients, due to overlapping conditions such as sepsis, CRS, and encephalitis.This case highlights the importance of institutional protocols, early recognition, and multidisciplinary management to improve outcomes.The rapid progression from ICANS 1 to 4 suggests that early, severe CRS may contribute to accelerated neurotoxicity.Further research is needed to refine risk stratification, identify biomarkers, and improve diagnostic tools, particularly in pediatric ICUs, to optimize CAR-T cell therapy outcomes.

Abstract licence: CC BY-NC-ND

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