CNS demyelination associated with immune dysregulation and a novel CLTA-4 variant

Stefania Kaninia*, Alexandros Grammatikos, Kathryn Urankar, Shelley Renowden, Nik Patel, Mark Gompels, Claire M Rice

*Corresponding author for this work

Research output: Contribution to journalLetter (Academic Journal)

Abstract

Background:
The cytotoxic T-lymphocyte antigen-4 (CTLA-4) pathway acts as a negative immune regulator of T cell activation and promotes self-tolerance.

Case:
We report the first case of biopsy-proven central nervous system inflammatory demyelination in the context of primary immunodeficiency and a novel CTLA-4 variant.

Conclusions:
This case has significant implications for the development of novel treatments for autoimmune conditions including Multiple Sclerosis and further emphasises the need for caution with clinical use of CTLA-4 immune checkpoint inhibitors in those with a history of inflammatory demyelination.


Original languageEnglish
JournalMultiple Sclerosis Journal
Publication statusAccepted/In press - 14 Sep 2020

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    Kaninia, S., Grammatikos, A., Urankar, K., Renowden, S., Patel, N., Gompels, M., & Rice, C. M. (Accepted/In press). CNS demyelination associated with immune dysregulation and a novel CLTA-4 variant. Multiple Sclerosis Journal.