A severe case of neuro-Sjögren's syndrome induced by pembrolizumab.
J Immunother Cancer
; 6(1): 110, 2018 10 22.
Article
em En
| MEDLINE
| ID: mdl-30348223
ABSTRACT
BACKGROUND:
The prevalence of connective tissue disease (CTD) induced by immune checkpoint inhibitors (CPIs) in the absence of pre-existing autoimmunity is unknown. CASE PRESENTATION 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.CONCLUSIONS:
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.Palavras-chave
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Base de dados:
MEDLINE
Assunto principal:
Síndrome de Sjogren
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Síndromes Neurotóxicas
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Anticorpos Monoclonais Humanizados
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Antineoplásicos Imunológicos
Tipo de estudo:
Risk_factors_studies
Limite:
Aged
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Female
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Humans
Idioma:
En
Ano de publicação:
2018
Tipo de documento:
Article