Evidence map›Paper›PMID 28536921›Full record

ReviewJournal of neurology2017

Progressive multifocal leukoencephalopathy in patients treated with fumaric acid esters: a review of 19 cases.

Robbert-Jan Gieselbach, Annemarie H Muller-Hansma, Martijn T Wijburg, Marjolein S de Bruin-Weller, Bob W van Oosten, Dennis J Nieuwkamp, Frank E Coenjaerts, Mike P Wattjes, Jean-Luc Murk

Erratum issuedAbstract readReview
PubMed Publisher
In one paragraph

Review in Journal of neurology, 2017. The graph could read no effect estimate from its abstract, so it casts no vote on the map. An erratum has been issued. Cited by 29 papers.

0numbers the graph read from it
0cells of the map it votes in
29citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

Neither the registry nor the abstract names a trial number. If this is a trial report, that itself is worth knowing.

3 · Its place in the literature

Who cites it

29 citing papers in PubMed.

  1. Trial
  2. Article
  3. Review
  4. Review
  5. Review
  6. [Low-dose, long-term fumaric acid esters in recalcitrant cutaneous sarcoidosis : Report of two cases].Der Hautarzt; Zeitschrift fur Dermatologie, Venerologie, und verwandte Gebiete · 2021
    Article
  7. Article
  8. Article
  9. Article
  10. Article
  11. Article
  12. Review
  13. Article
  14. Fumaric acid ester-induced T-cell lymphopenia in the real-life treatment of psoriasis.Journal of the European Academy of Dermatology and Venereology : JEADV · 2019
    Observational
  15. Article
  16. Review
  17. Review
  18. Observational
  19. Article
  20. Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

9 authors.

Robbert-Jan GieselbachDepartment of Medical Microbiology and Infection Control, University Medical Center Utrecht, Utrecht, The Netherlands.
Annemarie H Muller-HansmaNetherlands Pharmacovigilance Centre Lareb, 's Hertogenbosch, The Netherlands.
Martijn T WijburgDepartment of Neurology, Neuroscience Amsterdam, VUmc MS Center Amsterdam, VU University Medical Center, Amsterdam, The Netherlands.
Marjolein S de Bruin-WellerDepartment of Dermatology and Allergology, University Medical Center Utrecht, Utrecht, The Netherlands.
Bob W van OostenDepartment of Neurology, Neuroscience Amsterdam, VUmc MS Center Amsterdam, VU University Medical Center, Amsterdam, The Netherlands.
Dennis J NieuwkampDepartment of Neurology, Jeroen Bosch Hospital, 's-Hertogenbosch, The Netherlands.
Frank E CoenjaertsDepartment of Medical Microbiology and Infection Control, University Medical Center Utrecht, Utrecht, The Netherlands.
Mike P WattjesDepartment of Radiology and Nuclear Medicine, Neuroscience Amsterdam, VUmc MS Center Amsterdam, VU University Medical Center, Amsterdam, The Netherlands.
Jean-Luc MurkLaboratory of Medical Microbiology and Immunology, St. Elisabeth Hospital Tilburg, Tilburg, The Netherlands. j.murk@etz.nl.

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

Progressive multifocal leukoencephalopathy (PML) is a rare and potentially fatal condition caused by a brain infection with JC polyomavirus (JCV). PML develops almost exclusively in immunocompromised patients and has recently been associated with use of fumaric acid esters (FAEs), or fumarates. We reviewed the literature and the Dutch and European pharmacovigilance databases in order to identify all available FAE-associated PML cases and distinguish possible common features among these patients. A total of 19 PML cases associated with FAE use were identified. Five cases were associated with FAE use for multiple sclerosis and 14 for psoriasis. Ten patients were male and nine were female. The median age at PML diagnosis was 59 years. The median duration of FAE therapy to PML symptom onset or appearance of first PML lesion on brain imaging was 31 months (range 6-110). In all cases a certain degree of lymphocytopenia was reported. The median duration of lymphocytopenia to PML symptom onset was 23 months (range 6-72). The median lymphocyte count at PML diagnosis was 414 cells/µL. CD4 and CD8 counts were reported in ten cases, with median cell count of 137 and 39 cells/µL, respectively. Three patients died (16% mortality). The association between occurrence of PML in patients with low CD4 and CD8 counts is reminiscent of PML cases in the HIV population and suggests that loss of T cells is the most important risk factor.

Indexed as

AdultAgedAged, 80 and overFemaleFumaratesHumansImage Processing, Computer-AssistedLeukoencephalopathy, Progressive MultifocalMagnetic Resonance ImagingMaleMiddle AgedTreatment OutcomeFumaratesFumaric acid estersJC virusMultiple sclerosisProgressive multifocal leukoencephalopathyPsoriasis

Identifiers

What OpenQuestion holds

Textmetadata
Read underepoch 390

Registered trials

None linked

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.