A severe case of neuro-Sjögren's syndrome induced by pembrolizumab.

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Version: Final published version
License: CC BY 4.0
Serval ID
serval:BIB_3E8CC11BAFBA
Type
Article: article from journal or magazin.
Publication sub-type
Case report (case report): feedback on an observation with a short commentary.
Collection
Publications
Institution
Title
A severe case of neuro-Sjögren's syndrome induced by pembrolizumab.
Journal
Journal for immunotherapy of cancer
Author(s)
Ghosn J., Vicino A., Michielin O., Coukos G., Kuntzer T., Obeid M.
ISSN
2051-1426 (Electronic)
ISSN-L
2051-1426
Publication state
Published
Issued date
22/10/2018
Peer-reviewed
Oui
Volume
6
Number
1
Pages
110
Language
english
Notes
Publication types: Journal Article
Publication Status: epublish
Abstract
The prevalence of connective tissue disease (CTD) induced by immune checkpoint inhibitors (CPIs) in the absence of pre-existing autoimmunity is unknown.
We report the case of a melanoma patient treated for 8 months with pembrolizumab who developed a subacute ataxic sensory neuronopathy (SNN), including a right trigeminal neuropathy. Salivary gland biopsy showed inflammatory changes suggestive of Sjögren's syndrome, while brain MRI revealed enhancement of the right trigeminal ganglia. A high level of protein and pleocytosis was found in the cerebrospinal fluid, with negative cultures. Nerve conduction studies revealed the absence of sensory nerve action potentials in the upper and lower limbs and reduced motor responses in the upper limbs, fulfilling criteria for SNN. Blood tests revealed an important inflammatory syndrome, hemolytic anemia, elevation of total IgG levels and the presence of ANA autoantibodies specific to anti-SSA (52 and 60 kd). All these elements were absent before the initiation of the treatment with pembrolizumab. Initially, there was a clinical response following intravenous frontline methylprednisone, but the subacute relapse required the introduction of second-line treatment with intravenous immunoglobulins and then rituximab, which led to a quick clinical improvement.
Herein, we describe the first case of a patient who developed a typical SNN as a complication of severe neuro-Sjögren's syndrome induced by pembrolizumab treatment.
Keywords
Checkpoint inhibitors, Immune-related adverse events, Neuro-Sjögren’s syndrome, PD-1, Pembrolizumab
Pubmed
Web of science
Open Access
Yes
Create date
05/11/2018 9:43
Last modification date
20/08/2019 13:35
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