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

King's College Hospital NHS Foundation Trust
Maidstone and Tunbridge Wells NHS Trust

Research output: Contribution to journal › Article › peer-review

7 Citations (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
Article number	578248
Journal	Journal of Neuroimmunology
Volume	385
DOIs	https://doi.org/10.1016/j.jneuroim.2023.578248
Publication status	Published - 15 Dec 2023
Externally published	Yes

Keywords

Idiopathic lymphocytopenia
John Cunningham Virus
Neuroinflammation
PD-1 Checkpoint Inhibitor
Pembrolizumab
Progressive multifocal leukoencephalopathy

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10.1016/j.jneuroim.2023.578248

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@article{ea7afe2be3a1410f8499ba563e78bcca,

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 = "Idiopathic lymphocytopenia, John Cunningham Virus, Neuroinflammation, PD-1 Checkpoint Inhibitor, Pembrolizumab, Progressive multifocal leukoencephalopathy",

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\}",

note = "Publisher Copyright: {\textcopyright} 2023 Elsevier B.V.",

year = "2023",

month = dec,

day = "15",

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

language = "English",

volume = "385",

journal = "Journal of Neuroimmunology",

issn = "0165-5728",

publisher = "Elsevier B.V.",

}

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 - Idiopathic lymphocytopenia

KW - John Cunningham Virus

KW - Neuroinflammation

KW - PD-1 Checkpoint Inhibitor

KW - Pembrolizumab

KW - Progressive multifocal leukoencephalopathy

UR - https://www.scopus.com/pages/publications/85177785549

U2 - 10.1016/j.jneuroim.2023.578248

DO - 10.1016/j.jneuroim.2023.578248

M3 - Article

C2 - 37995595

AN - SCOPUS:85177785549

SN - 0165-5728

VL - 385

JO - Journal of Neuroimmunology

JF - Journal of Neuroimmunology

M1 - 578248

ER -

Progressive multifocal leukoencephalopathy secondary to idiopathic CD4 lymphocytopenia treated with pembrolizumab

Abstract

Keywords

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