CAR T-cell-associated acute myelopathy: a rare but critical neurologic toxicity to recognize

Chimeric Antigen Receptor T-cell (CAR-T) therapy has significantly improved outcomes in hematologic malignancies but may lead to rare and severe neurological complications beyond immune effector cell-associated neurotoxicity syndrome (ICANS), such as acute myelopathy. We report the case of a 59-year-old woman treated with CD19-targeted CAR-T cells for relapsed lymphoma, who developed grade IV ICANS followed by a clinical presentation of myelitis attributed to CAR T-cell therapy after exclusion of alternative diagnoses. Aggressive immunosuppressive treatment and supportive care failed to achieve neurological recovery. The patient remained ventilator-dependent due to cervical spinal cord lesions and ultimately died following transition to palliative care. This case underscores the diagnostic challenges posed by this medical emergency in critically ill patients since numerous confounding factors may obscure early signs of spinal cord involvement in the ICU setting. Early neurologic evaluation and multidisciplinary management are critical. This case aims to raise awareness on this rare yet dramatic complication, in which prompt initiation of treatment may prevent long term sequelae.