Diabetes insipidus and Guillain-Barré-like syndrome following CAR-T cell therapy: a case report

BackgroundImmune effector cell-associated neurotoxicity syndrome (ICANS) is a common adverse event of CD19-directed chimeric antigen receptor (CAR) T cell therapy. Other neurological adverse events, however, have not methodically been described and studied. Furthermore, safety data on CAR-T cell the...

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Bibliographic Details
Published in:Journal for immunotherapy of cancer Vol. 11; no. 1; p. e006059
Main Authors: Koch, Christian, Fleischer, Juliane, Popov, Todor, Frontzek, Karl, Schreiner, Bettina, Roth, Patrick, Manz, Markus G., Unseld, Simone, Müller, Antonia M. S., Russkamp, Norman F.
Format: Journal Article
Language:English
Published: England BMJ Publishing Group Ltd 01.01.2023
BMJ Publishing Group LTD
BMJ Publishing Group
Subjects:
Antigens
Blood cancer
Cancer
Case Report
Case reports
Cell- and Tissue-Based Therapy
Cells
Central Nervous System Neoplasms
Cytokines
Diabetes
Diabetes Insipidus - etiology
Diabetes Mellitus
Dyspnea
Guillain-Barre syndrome
hematologic neoplasms
Humans
Hypothalamus
Immunoglobulins
Immunotherapy
Immunotherapy, Adoptive - adverse effects
Lymphocytes
Lymphoma
Lymphoma, Non-Hodgkin
Nervous system
Neurological disorders
Neurotoxicity
Ostomy
Pathophysiology
Pituitary gland
Proteins
Receptors, Chimeric Antigen
Urine
Ventilators
hematologic neoplasms
case reports
receptors, chimeric antigen
immunotherapy
lymphoma
ISSN:2051-1426, 2051-1426
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Summary:BackgroundImmune effector cell-associated neurotoxicity syndrome (ICANS) is a common adverse event of CD19-directed chimeric antigen receptor (CAR) T cell therapy. Other neurological adverse events, however, have not methodically been described and studied. Furthermore, safety data on CAR-T cell therapy in patients with central nervous system (CNS) lymphoma remain limited.Main bodyWe here report occurrence of a Guillain-Barré-like syndrome (GBS) and central diabetes insipidus (cDI) following tisagenlecleucel therapy for relapsed high-grade lymphoma with CNS involvement. Both complications were refractory to standard treatment of ICANS. Weakness of respiratory muscles required mechanical ventilation and tracheostomy while cDI was treated with desmopressin substitution for several weeks. Muscle-nerve biopsy and nerve conduction studies confirmed an axonal pattern of nerve damage. T cell-rich infiltrates and detection of the CAR transgene in muscle-nerve sections imply a direct or indirect role of CAR-T cell-mediated inflammation. In line with current treatment guidelines for GBS, intravenous immunoglobulin was administered and gradual but incomplete recovery was observed over the course of several months.ConclusionsThis case report highlights the risk of rare but severe neurological adverse events, such as acute GBS or cDI, in patients treated with CAR-T cells. It further underlines the importance of appropriate patient surveillance and systematic reporting of rare complications to eventually improve treatment.
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ISSN:2051-1426
2051-1426
DOI:10.1136/jitc-2022-006059