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Case Reports
. 2020 Oct 23;1(2):585-588.
doi: 10.1002/jha2.56. eCollection 2020 Nov.

Improvement in progressive multifocal leukoencephalopathy after pembrolizumab-induced immune reconstruction inflammatory syndrome in a patient with follicular lymphoma

Affiliations
Case Reports

Improvement in progressive multifocal leukoencephalopathy after pembrolizumab-induced immune reconstruction inflammatory syndrome in a patient with follicular lymphoma

Inès Dufour et al. EJHaem. .

Abstract

Progressive multifocal leukoencephalopathy (PML) may develop in follicular lymphoma patients treated with bendamustine-rituximab. In this report, treatment with pembrolizumab successfully inhibited the clinical progression of PML by promoting radiologically demonstrated immune restoration inflammatory syndrome (IRIS), allowing complete clearance of the virus. These findings may further support the use of pembrolizumab in PML with special consideration for the potential occurrence of IRIS.

Keywords: Follicular lymphoma; JC virus; immune reconstruction inflammatory syndrome; pembrolizumab; progressive multifocal leukoencephalopathy.

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Conflict of interest statement

The authors declare no conflict of interest.Inès Dufour, Eric Van Den Neste, Thierry Duprez, Marie Wertz, Pascale Saussoy, Nathalie Ackermans, Souraya El Sankari, and Vincent van Pesch contributed to patient management, data collection, and revised the paper. Inès Dufour and Eric Van Den Neste wrote the paper.

Figures

FIGURE 1
FIGURE 1
Brain magnetic resonance monitoring (all images in similar slice location). Upper row: Contrast‐enhanced FLAIR (fluid‐attenuated inversion recovery) views. Lower row: Contrast‐enhanced T1‐weighted views. A and B, Pretreatment baseline examination showing bilateral but asymmetric hyperintense changes in the parietal white matter on FLAIR image (A) appearing hypointense with only a few enhanced foci on post‐contrast image (B). C and D, Follow‐up examination 5 weeks later showing increase in size of damaged parenchyma on FLAIR image (C) together with dramatic increase of contrast‐enhancement featuring immune restoration (D). E and F, Follow‐up examination 11 weeks later showing moderate subsidence of FLAIR abnormalities (E) but strong decrease of areas of contrast enhancement (F) featuring the restoration of a competent brain‐blood barrier (BBB) after the inflammatory blaze seen in (D)

References

    1. D'Alo F, Malafronte R, Piludu F, Bellesi S, Cuccaro A, Maiolo E, et al. Progressive multifocal leukoencephalopathy in patients with follicular lymphoma treated with bendamustine plus rituximab followed by rituximab maintenance. Br J Haematol. 2020;189:e140‐4. - PubMed
    1. Brice P, Bastion Y, Lepage E, Brousse N, Haioun C, Moreau P, et al. Comparison in low‐tumor‐burden follicular lymphomas between an initial no‐treatment policy, prednimustine, or interferon alfa: a randomized study from the Groupe d'Etude des Lymphomes Folliculaires. Groupe d'Etude des Lymphomes de l'Adulte. J Clin Oncol. 1997;15:1110‐7. - PubMed
    1. Neil EC, DeAngelis LM. Progressive multifocal leukoencephalopathy and hematologic malignancies: a single cancer center retrospective review. Blood Adv. 2017;1:2041‐5. - PMC - PubMed
    1. Major EO. Progressive multifocal leukoencephalopathy in patients on immunomodulatory therapies. Annu Rev Med. 2010;61:35‐47. - PubMed
    1. Du Pasquier RA, Clark KW, Smith PS, Joseph JT, Mazullo JM, De Girolami U, et al. JCV‐specific cellular immune response correlates with a favorable clinical outcome in HIV‐infected individuals with progressive multifocal leukoencephalopathy. J Neurovirol. 2001;7:318–22. - PubMed

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