Autonomic dysfunction as manifestation of ICANS: A case report

Medicine (Baltimore). 2024 Sep 6;103(36):e38659. doi: 10.1097/MD.0000000000038659.

Abstract

Rationale: Anti-CD19 chimeric antigen receptor T-cell (CAR-T) therapy is a successful treatment for B-cell malignancies associated with cytokine release syndrome (CRS) and immune effector cell-associated neurotoxicity syndrome (ICANS). Cardiovascular toxicities have also been reported in this setting. However, there is scarce data regarding development of autonomic disorders after CAR-T cell therapy.

Patient concerns: We report a case with a patient with non-Hodgkin B-cell lymphoma, refractory to 2 prior lines of immunochemotherapy, treated with CAR-T therapy.

Diagnoses: Orthostatic hypotension secondary to autonomic dysfunction was diagnosed as manifestation of ICANS.

Interventions: The patient received metilprednisolone 1000 mg IV daily for 3 days and anakinra 100 mg IV every 6h.

Outcomes: The vast majority of autonomic symptoms ceased and 4 months after CAR-T therapy, autonomic dysfunction was resolved.

Lessons: New-onset autonomic dysfunction can occur as manifestation of ICANS in patients who experience persistent neurologic and cardiovascular symptoms after resolution of acute neurotoxicity and should be early recognized. Differences in differential diagnosis, mechanisms and treatment approaches are discussed.

Publication types

  • Case Reports

MeSH terms

  • Autonomic Nervous System Diseases* / diagnosis
  • Autonomic Nervous System Diseases* / etiology
  • Cytokine Release Syndrome / etiology
  • Humans
  • Hypotension, Orthostatic / diagnosis
  • Hypotension, Orthostatic / etiology
  • Immunotherapy, Adoptive / adverse effects
  • Lymphoma, B-Cell / complications
  • Lymphoma, B-Cell / drug therapy
  • Male
  • Methylprednisolone / therapeutic use
  • Middle Aged
  • Neurotoxicity Syndromes / diagnosis
  • Neurotoxicity Syndromes / etiology

Substances

  • Methylprednisolone