Progressive multifocal leukoencephalopathy secondary to idiopathic CD4 lymphocytopenia treated with pembrolizumab

Kyriakoula Varmpompiti; Andrew J Westwood; Aaron Ben-Joseph; Naomi Sibtain; Mohammad A A Ibrahim; Biba Stanton; Mark Zuckerman; Robert Hadden; Laura Mantoan Ritter

doi:10.1016/j.jneuroim.2023.578248

Progressive multifocal leukoencephalopathy secondary to idiopathic CD4 lymphocytopenia treated with pembrolizumab

Kyriakoula Varmpompiti, Andrew J Westwood, Aaron Ben-Joseph, Naomi Sibtain, Mohammad A A Ibrahim, Biba Stanton, Mark Zuckerman, Robert Hadden, Laura Mantoan Ritter

Peter Gorer Department of Immunobiology

Research output: Contribution to journal › Review article › peer-review

1 Citation (Scopus)

Abstract

BACKGROUND: Progressive multifocal leukoencephalopathy (PML) is a rare demyelinating disease due to a lytic infection of oligodendrocytes caused by John Cunningham polyoma virus (JCV) infection. Idiopathic CD4+ T-cell lymphocytopenia (ICL) is a very rare cause of PML.

METHODS: We present an individual with PML secondary to ICL treated with 3 doses of pembrolizumab, a Programmed-Death-1 Immune Checkpoint Inhibitor following with complete resolution of symptoms and conduct a review of the literature.

CONCLUSION: This report illustrates the objective clinical and radiological improvement in a patient with PML due to ICL and suggests further study of immune checkpoint inhibitors as potential treatment for patients with PML.

Original language	English
Pages (from-to)	578248
Journal	Journal of Neuroimmunology
Volume	385
DOIs	https://doi.org/10.1016/j.jneuroim.2023.578248
Publication status	Published - 15 Dec 2023

Keywords

Humans
Leukoencephalopathy, Progressive Multifocal/diagnostic imaging
T-Lymphocytopenia, Idiopathic CD4-Positive/complications
Antibodies, Monoclonal, Humanized/therapeutic use
JC Virus

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10.1016/j.jneuroim.2023.578248

Cite this

@article{4ebe871bdc5a4636ae3ba40fdeb72f3f,

title = "Progressive multifocal leukoencephalopathy secondary to idiopathic CD4 lymphocytopenia treated with pembrolizumab",

abstract = "BACKGROUND: Progressive multifocal leukoencephalopathy (PML) is a rare demyelinating disease due to a lytic infection of oligodendrocytes caused by John Cunningham polyoma virus (JCV) infection. Idiopathic CD4+ T-cell lymphocytopenia (ICL) is a very rare cause of PML.METHODS: We present an individual with PML secondary to ICL treated with 3 doses of pembrolizumab, a Programmed-Death-1 Immune Checkpoint Inhibitor following with complete resolution of symptoms and conduct a review of the literature.CONCLUSION: This report illustrates the objective clinical and radiological improvement in a patient with PML due to ICL and suggests further study of immune checkpoint inhibitors as potential treatment for patients with PML.",

keywords = "Humans, Leukoencephalopathy, Progressive Multifocal/diagnostic imaging, T-Lymphocytopenia, Idiopathic CD4-Positive/complications, Antibodies, Monoclonal, Humanized/therapeutic use, JC Virus",

author = "Kyriakoula Varmpompiti and Westwood, {Andrew J} and Aaron Ben-Joseph and Naomi Sibtain and Ibrahim, {Mohammad A A} and Biba Stanton and Mark Zuckerman and Robert Hadden and Ritter, {Laura Mantoan}",

year = "2023",

month = dec,

day = "15",

doi = "10.1016/j.jneuroim.2023.578248",

language = "English",

volume = "385",

pages = "578248",

journal = "Journal of Neuroimmunology",

issn = "0165-5728",

publisher = "Elsevier",

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TY - JOUR

T1 - Progressive multifocal leukoencephalopathy secondary to idiopathic CD4 lymphocytopenia treated with pembrolizumab

AU - Varmpompiti, Kyriakoula

AU - Westwood, Andrew J

AU - Ben-Joseph, Aaron

AU - Sibtain, Naomi

AU - Ibrahim, Mohammad A A

AU - Stanton, Biba

AU - Zuckerman, Mark

AU - Hadden, Robert

AU - Ritter, Laura Mantoan

PY - 2023/12/15

Y1 - 2023/12/15

N2 - BACKGROUND: Progressive multifocal leukoencephalopathy (PML) is a rare demyelinating disease due to a lytic infection of oligodendrocytes caused by John Cunningham polyoma virus (JCV) infection. Idiopathic CD4+ T-cell lymphocytopenia (ICL) is a very rare cause of PML.METHODS: We present an individual with PML secondary to ICL treated with 3 doses of pembrolizumab, a Programmed-Death-1 Immune Checkpoint Inhibitor following with complete resolution of symptoms and conduct a review of the literature.CONCLUSION: This report illustrates the objective clinical and radiological improvement in a patient with PML due to ICL and suggests further study of immune checkpoint inhibitors as potential treatment for patients with PML.

AB - BACKGROUND: Progressive multifocal leukoencephalopathy (PML) is a rare demyelinating disease due to a lytic infection of oligodendrocytes caused by John Cunningham polyoma virus (JCV) infection. Idiopathic CD4+ T-cell lymphocytopenia (ICL) is a very rare cause of PML.METHODS: We present an individual with PML secondary to ICL treated with 3 doses of pembrolizumab, a Programmed-Death-1 Immune Checkpoint Inhibitor following with complete resolution of symptoms and conduct a review of the literature.CONCLUSION: This report illustrates the objective clinical and radiological improvement in a patient with PML due to ICL and suggests further study of immune checkpoint inhibitors as potential treatment for patients with PML.

KW - Humans

KW - Leukoencephalopathy, Progressive Multifocal/diagnostic imaging

KW - T-Lymphocytopenia, Idiopathic CD4-Positive/complications

KW - Antibodies, Monoclonal, Humanized/therapeutic use

KW - JC Virus

U2 - 10.1016/j.jneuroim.2023.578248

DO - 10.1016/j.jneuroim.2023.578248

M3 - Review article

C2 - 37995595

SN - 0165-5728

VL - 385

SP - 578248

JO - Journal of Neuroimmunology

JF - Journal of Neuroimmunology

ER -

Progressive multifocal leukoencephalopathy secondary to idiopathic CD4 lymphocytopenia treated with pembrolizumab

Abstract

Keywords

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