For Publishers
DiscoveryMetadataPeer reviewHostingPublishing
For Researchers
JoinPublishReviewCollect
Register
Blog
About
Search
Advanced search
My ScienceOpen
Search
For Publishers
For Researchers
Blog
About
7
views
8
references
 Top references
cited by
4
    
0 reviews
 Review
0
comments
 Comment
0
recommends
+1 Recommend
0 collections
    0
    shares
      282
      similar
       All similar
      • Record: found
      • Abstract: found
      • Article: found
      Is Open Access

      Extensive immune reconstitution inflammatory syndrome in Fingolimod-associated PML: a case report with 7 Tesla MRI data

      case-report

      Read this article at

      ScienceOpenPublisherPMC
      Bookmark
          There is no author summary for this article yet. Authors can add summaries to their articles on ScienceOpen to make them more accessible to a non-specialist audience.

          Abstract

          Background

          Progressive multifocal leukoencephalopathy (PML) is a rare complication of patients treated with fingolimod.

          Case presentation

          Routine MRI eventually led to diagnosis of asymptomatic early PML that remained stable after discontinuation of fingolimod. As blood lymphocyte counts normalized, signs of immune reconstitution inflammatory syndrome (IRIS) and renewed MS activity developed. Both, advanced laboratory and ultrahigh field MRI findings elucidated differences between PML and MS.

          Conclusions

          In our case, early discontinuation of fingolimod yielded a good outcome, lymphocyte counts reflected immune system activity, and paraclinical findings helped to differentiate between PML-IRIS and MS.

          Related collections

          Most cited references8

          • Record: found
          • Abstract: found
          • Article: not found

          Dimethyl fumarate selectively reduces memory T cells in multiple sclerosis patients.

          EE Longbrake, MJ Ramsbottom, C Cantoni … (2016)
          Dimethyl fumarate (DMF) alters the phenotype of circulating immune cells and causes lymphopenia in a subpopulation of treated multiple sclerosis (MS) patients.
          Article 0 comments Cited 51 times – based on 0 reviews     Review now
            Bookmark
            • Record: found
            • Abstract: not found
            • Article: not found

            Progressive multifocal leukoencephalopathy after fingolimod treatment

            Bernhard Hemmer, Bruce A C Cree, Benjamin Greenberg … (2018)
            Article 0 comments Cited 49 times – based on 0 reviews     Review now
              Bookmark
              • Record: found
              • Abstract: found
              • Article: not found

              Fatal PML associated with efalizumab therapy: insights into integrin αLβ2 in JC virus control.

              Fabian Schneider, R Ransohoff, W. Brück … (2012)
              Progressive multifocal leukoencephalopathy (PML) has become much more common with monoclonal antibody treatment for multiple sclerosis and other immune-mediated disorders. We report 2 patients with severe psoriasis and fatal PML treated for ≥3 years with efalizumab, a neutralizing antibody to αLβ2-leukointegrin (LFA-1). In one patient, we conducted serial studies of peripheral blood and CSF including analyses of leukocyte phenotypes, migration ex vivo, and CDR3 spectratypes with controls coming from HIV-infected patients with PML. Extensive pathologic and histologic analysis was done on autopsy CNS tissue of both patients. Both patients developed progressive cognitive and motor deficits, and JC virus was identified in CSF. Despite treatment including plasma exchange (PE) and signs of immune reconstitution, both died of PML 2 and 6 months after disease onset. Neuropathologic examination confirmed PML. Efalizumab treatment was associated with reduced transendothelial migration by peripheral T cells in vitro. As expression levels of LFA-1 on peripheral T cells gradually rose after PE, in vitro migration increased. Peripheral and CSF T-cell spectratyping showed CD8+ T-cell clonal expansion but blunted activation, which was restored after PE. From these data we propose that inhibition of peripheral and intrathecal T-cell activation and suppression of CNS effector-phase migration both characterize efalizumab-associated PML. LFA-1 may be a crucial factor in homeostatic JC virus control.
              Article 0 comments Cited 31 times – based on 0 reviews     Review now
                Bookmark
                All references

                Author and article information

                Contributors
                Tim Sinnecker: tim.sinnecker@miac.ch
                Jeffrie Hadisurya: jeffrie.hadisurya@krupp-krankenhaus.de
                Tilman Schneider-Hohendorf: tilman.schneider-hohendorf@ukmuenster.de
                Nicholas Schwab: nicholas.schwab@ukmuenster.de
                Karsten Wrede: Karsten.Wrede@uk-essen.de
                Oliver Gembruch: oliver.gembruch@uk-essen.de
                Ralf Gold: Ralf.Gold@rub.de
                Kerstin Hellwig: k.hellwig@klinikum-bochum.de
                Sara Pilgram-Pastor: saramagdalena.pilgram-pastor@krupp-krankenhaus.de
                Ortwin Adams: ortwin.adams@uni-duesseldorf.de
                Philipp Albrecht: phil.albrecht@googlemail.com
                Hans-Peter Hartung: hans-peter.hartung@uni-duesseldorf.de
                Orhan Aktas: orhan.aktas@uni-duesseldorf.de
                Markus Kraemer:
                ORCID: http://orcid.org/0000-0002-5667-3621
                +49-201-434-41-422 , markus.kraemer@krupp-krankenhaus.de
                Journal
                Journal ID (nlm-ta): BMC Neurol
                Journal ID (iso-abbrev): BMC Neurol
                Title: BMC Neurology
                Publisher: BioMed Central (London )
                ISSN (Electronic): 1471-2377
                Publication date (Electronic): 9 August 2019
                Publication date PMC-release: 9 August 2019
                Publication date Collection: 2019
                Volume: 19
                Electronic Location Identifier: 190
                Affiliations
                [1 ]GRID grid.410567.1, Department of Neurology, , Universitätsspital, ; Basel, Switzerland
                [2 ]Medical Image Analysis Center Basel, Basel, Switzerland
                [3 ]Department of Neurology, Alfried Krupp von Bohlen und Halbach Hospital, Alfried-Krupp-Str. 21, 45117 Essen, Germany
                [4 ]ISNI 0000 0004 0551 4246, GRID grid.16149.3b, Clinic of Neurology with Institute of Translational Neurology, , University Hospital Münster, ; Münster, Germany
                [5 ]Department of Neurosurgery, University Hospital Essen, University Duisburg-Essen, Essen, Germany
                [6 ]ISNI 0000 0001 2187 5445, GRID grid.5718.b, Erwin L. Hahn Institute for Magnetic Resonance Imaging, , University Duisburg-Essen, ; Essen, Germany
                [7 ]ISNI 0000 0004 0490 981X, GRID grid.5570.7, Department of Neurology, St. Josef Hospital, , Ruhr-University Bochum, ; Bochum, Germany
                [8 ]GRID grid.476313.4, Department of Neuroradiology, , Alfried Krupp Hospital, ; Essen, Germany
                [9 ]ISNI 0000 0001 2176 9917, GRID grid.411327.2, Institute of Virology, Medical Faculty, , Heinrich-Heine University Düsseldorf, ; Düsseldorf, Germany
                [10 ]ISNI 0000 0001 2176 9917, GRID grid.411327.2, Department of Neurology, Medical Faculty, , Heinrich-Heine University Düsseldorf, ; Düsseldorf, Germany
                Author information
                http://orcid.org/0000-0002-5667-3621
                Article
                Publisher ID: 1407
                DOI: 10.1186/s12883-019-1407-2
                PMC ID: 6688281
                PubMed ID: 31399069
                SO-VID: 84a6850e-8217-4735-a165-b31e94fe9b52
                Copyright © © The Author(s). 2019
                License:

                Open AccessThis article is distributed under the terms of the Creative Commons Attribution 4.0 International License ( http://creativecommons.org/licenses/by/4.0/), which permits unrestricted use, distribution, and reproduction in any medium, provided you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons license, and indicate if changes were made. The Creative Commons Public Domain Dedication waiver ( http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated.

                History
                Date received : 9 March 2019
                Date accepted : 19 July 2019
                Categories
                Subject: Case Report
                Custom metadata
                issue-copyright-statement © The Author(s) 2019

                ScienceOpen disciplines: Neurology
                Keywords: progressive multifocal leucencephalopathy,immune reconstitution inflammatory syndrome,fingolimod,multiple sclerosis,7 tesla mri
                Data availability:
                ScienceOpen disciplines: Neurology
                Keywords: progressive multifocal leucencephalopathy, immune reconstitution inflammatory syndrome, fingolimod, multiple sclerosis, 7 tesla mri

                Comments

                Comment on this article

                scite_

                Similar content282

                See all similar

                Cited by4

                See all cited by

                Most referenced authors135

                See all reference authors