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Published on: September 25, 2018
Progressive multifocal leukoencephalopathy secondary to idiopathic CD4 lymphocytopenia treated with pembrolizumab
Kyriakoula Varmpompiti1, Andrew J Westwood2, Aaron Ben-Joseph2
1King's College Hospital NHS Foundation Trust, London, United Kingdom.
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.
Insights
Idiopathic CD4+ T-cell lymphocytopenia (ICL) is a rare cause of progressive multifocal leukoencephalopathy (PML). Treatment with pembrolizumab, a Programmed-Death-1 Immune Checkpoint Inhibitor, led to complete symptom resolution in one patient.
Area of Science:
- Neuroimmunology
- Viral Pathogenesis
Background:
- Progressive multifocal leukoencephalopathy (PML) is a rare demyelinating disease caused by John Cunningham polyoma virus (JCV) infection of oligodendrocytes.
- Idiopathic CD4+ T-cell lymphocytopenia (ICL) is an exceedingly rare condition that can precipitate PML.
- Understanding the interplay between immune deficiency and opportunistic infections like PML is crucial for clinical management.
Approach:
- A case report detailing a patient with PML secondary to ICL.
- The patient received three doses of pembrolizumab, a Programmed-Death-1 Immune Checkpoint Inhibitor.
- A comprehensive literature review was conducted to contextualize this case.
Key Points:
- Successful treatment of PML secondary to ICL with pembrolizumab.
- Demonstrated objective clinical and radiological improvement post-treatment.
- Highlights the potential of immune checkpoint inhibitors in managing PML.
Conclusions:
- Immune checkpoint inhibitors, such as pembrolizumab, show promise as a therapeutic strategy for PML.
- Further research is warranted to explore the efficacy and safety of these agents in PML patients.
- This case underscores the importance of considering rare immune deficiencies in the etiology of PML.
