Skip to main content
Top
Published in: BMC Neurology 1/2022

Open Access 01-12-2022 | Autopsy | Case report

Toxic leukoencephalopathy with axonal spheroids caused by chemotherapeutic drugs other than methotrexate

Authors: Ka Young Lim, Seong-Ik Kim, Hyunhee Kim, Jeongwan Kang, Jin Woo Park, Jae Kyung Won, Dong-Yeop Shin, Sung-Hye Park

Published in: BMC Neurology | Issue 1/2022

Login to get access

Abstract

Background

The objective of this report is to share the clinicopathological features of chemotherapy-induced toxic leukoencephalopathy, which is a rare and under-recognized disease, clinically characterized by rapidly progressive cognitive loss that often leads to sudden death.

Case presentation

A 64-year-old woman and a 63-year-old man, who had both suffered from a rapid deterioration of consciousness, were autopsied under the clinical impressions of either the central nervous system graft versus host disease (CNS-GVHD), infectious encephalitis, or autoimmune encephalitis. Both patients had been treated with multiple chemotherapy regimens, including adriamycin, cytarabine arabinoside, daunorubicin, fludarabine, azacitidine, and allogeneic peripheral blood stem cell transplantation to treat hematological malignancies (acute myelogenous leukemia and myelodysplastic syndrome). Neuropathological findings at autopsy revealed rarefaction and vacuolar changes of the white matter with axonal spheroids, reactive gliosis, and foamy macrophage infiltration, predominantly in the visual pathways of the occipital and temporal lobes. Damaged axons exhibited immunoreactivity to beta-amyloid, consistent with axonopathy. However, there was no lymphocyte infiltration that suggested CNS-GVHD or any type of encephalitis.

Conclusion

The neuropathology found in the presented cases had the characteristic features of toxic leukoencephalopathy (chemobrain). Our cases showed that toxic leukoencephalopathy can also be caused by chemotherapy drugs other than methotrexate.
Literature
7.
go back to reference Schlegel U. Central Nervous System Toxicity of Chemotherapy. Eur Assoc NeuroOncoly Magazine. 2011;1(1):25–9. Schlegel U. Central Nervous System Toxicity of Chemotherapy. Eur Assoc NeuroOncoly Magazine. 2011;1(1):25–9.
9.
go back to reference Mahadeo KM, Dhall G, Panigrahy A, Lastra C, Ettinger LJ. Subacute methotrexate neurotoxicity and cerebral venous sinus thrombosis in a 12-year-old with acute lymphoblastic leukemia and methylenetetrahydrofolate reductase (MTHFR) C677T polymorphism: homocysteine-mediated methotrexate neurotoxicity via direct endothelial injury. Pediatr Hematol Oncol. 2010;27(1):46–52. https://doi.org/10.3109/08880010903341904.CrossRefPubMed Mahadeo KM, Dhall G, Panigrahy A, Lastra C, Ettinger LJ. Subacute methotrexate neurotoxicity and cerebral venous sinus thrombosis in a 12-year-old with acute lymphoblastic leukemia and methylenetetrahydrofolate reductase (MTHFR) C677T polymorphism: homocysteine-mediated methotrexate neurotoxicity via direct endothelial injury. Pediatr Hematol Oncol. 2010;27(1):46–52. https://​doi.​org/​10.​3109/​0888001090334190​4.CrossRefPubMed
14.
go back to reference Schlegel U. Central Nervous System Toxicity of Chemotherapy. Eur Assoc NeuroOncoly Magazine. 2011;1(1):25–9. Schlegel U. Central Nervous System Toxicity of Chemotherapy. Eur Assoc NeuroOncoly Magazine. 2011;1(1):25–9.
16.
go back to reference Pavletic ZS, Bishop MR, Markopoulou K, Wszolek ZK. Drug-induced parkinsonism after allogeneic bone marrow transplantation. Bone Marrow Transplant. 1996;17(6):1185–7.PubMed Pavletic ZS, Bishop MR, Markopoulou K, Wszolek ZK. Drug-induced parkinsonism after allogeneic bone marrow transplantation. Bone Marrow Transplant. 1996;17(6):1185–7.PubMed
Metadata
Title
Toxic leukoencephalopathy with axonal spheroids caused by chemotherapeutic drugs other than methotrexate
Authors
Ka Young Lim
Seong-Ik Kim
Hyunhee Kim
Jeongwan Kang
Jin Woo Park
Jae Kyung Won
Dong-Yeop Shin
Sung-Hye Park
Publication date
01-12-2022
Publisher
BioMed Central
Published in
BMC Neurology / Issue 1/2022
Electronic ISSN: 1471-2377
DOI
https://doi.org/10.1186/s12883-022-02818-8

Other articles of this Issue 1/2022

BMC Neurology 1/2022 Go to the issue