| Autor: |
Xavier, Boumaza, Baptiste, Bonneau, Damien, Roos-Weil, Carmela, Pinnetti, Sebastian, Rauer, Louisa, Nitsch, Arnaud, Del Bello, Ilijas, Jelcic, Kurt-Wolfram, Sühs, Jacques, Gasnault, Yasemin, Goreci, Oliver, Grauer, Sharmilee, Gnanapavan, Rebecca, Wicklein, Nicolas, Lambert, Thomas, Perpoint, Martijn, Beudel, David, Clifford, Agnès, Sommet, Irene, Cortese, Guillaume, Martin-Blondel |
| Rok vydání: |
2022 |
| Předmět: |
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| Zdroj: |
Annals of neurologyReferences. |
| ISSN: |
1531-8249 |
| Popis: |
Our aim was to assess the real-world effectiveness of immune checkpoint inhibitors for treatment of patients with progressive multifocal leukoencephalopathy (PML).We conducted a multicenter survey compiling retrospective data from 79 PML patients, including 38 published cases and 41 unpublished cases, who received immune checkpoint inhibitors as add-on to standard of care. One-year follow-up data were analyzed to determine clinical outcomes and safety profile. Logistic regression was used to identify variables associated with 1-year survival.Predisposing conditions included hematological malignancy (n = 38, 48.1%), primary immunodeficiency (n = 14, 17.7%), human immunodeficiency virus/acquired immunodeficiency syndrome (n = 12, 15.2%), inflammatory disease (n = 8, 10.1%), neoplasm (n = 5, 6.3%), and transplantation (n = 2, 2.5%). Pembrolizumab was most commonly used (n = 53, 67.1%). One-year survival was 51.9% (41/79). PML-immune reconstitution inflammatory syndrome (IRIS) was reported in 15 of 79 patients (19%). Pretreatment expression of programmed cell death-1 on circulating T cells did not differ between survivors and nonsurvivors. Development of contrast enhancement on follow-up magnetic resonance imaging at least once during follow-up (OR = 3.16, 95% confidence interval = 1.20-8.72, p = 0.02) was associated with 1-year survival. Cerebrospinal fluid JC polyomavirus DNA load decreased significantly by 1-month follow-up in survivors compared to nonsurvivors (p 0.0001). Thirty-two adverse events occurred among 24 of 79 patients (30.4%), and led to treatment discontinuation in 7 of 24 patients (29.1%).In this noncontrolled retrospective study of patients with PML who were treated with immune checkpoint inhibitors, mortality remains high. Development of inflammatory features or overt PML-IRIS was commonly observed. This study highlights that use of immune checkpoint inhibitors should be strictly personalized toward characteristics of the individual PML patient. ANN NEUROL 2022. |
| Databáze: |
OpenAIRE |
| Externí odkaz: |
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