Chimeric antigen receptor (CAR) T-cell therapy has revolutionized the treatment of patients with relapsed or refractory hematologic malignancies, but it comes with unique toxicities, notably cytokine release syndrome (CRS) and immune effector cell-associated neurotoxicity syndrome (ICANS). As experience with CAR T-cell therapy grows, distinct and infrequent neurological complications are becoming increasingly evident. Recently, reports of acute myelopathy following the administration of CAR T-cell therapies have been accumulating. Despite the establishment of consensus guidelines for managing ICANS, there remains limited guidance on appropriate investigations and treatments for this rare complication. Here, we delve into the clinical features, pathophysiology, and strategies for optimally managing acute myelitis after CAR T-cell therapy, drawing insights from reported cases in the literature.

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