Robert Hoepner1, Eva M Kolb1, Stefanie Dahlhaus2, Kerstin Hellwig1, Ortwin Adams3, Ingo Kleiter1, Anke Salmen4, Ruth Schneider1, Carsten Lukas5, Andrew Chan4, Joseph R Berger6, Ralf Gold1. 1. Department of Neurology, St. Josef Hospital, Ruhr University Bochum, Bochum, Germany. 2. Department of Neurology, Alfried Krupp Hospital, Essen, Germany. 3. Department of Virology, Heinrich Heine University, Düsseldorf, Germany. 4. Department of Neurology, St. Josef Hospital, Ruhr University Bochum, Bochum, Germany/Department of Neurology, University Hospital Bern and University of Bern, Bern, Switzerland. 5. Department of Radiology, St. Josef Hospital, Ruhr University Bochum, Bochum, Germany. 6. Department of Neurology, University of Pennsylvania, Philadelphia, PA, USA.
Abstract
OBJECTIVE: Progressive multifocal leukoencephalopathy (PML) is an emerging complication of immunosuppressive therapies, especially natalizumab in multiple sclerosis (MS). Factors associated with functional outcome of natalizumab-associated PML (natalizumab-PML) have not been sufficiently described. METHODS: We retrospectively analyzed medical records of all patients with natalizumab-PML ( n = 32) treated in our hospital since 2009. Disability measured by Expanded Disability Status Scale (EDSS) at two different time points (highest available EDSS during PML and last available EDSS after PML diagnosis) served as functional outcome parameters. Clinical, laboratory, and imaging data were analyzed for association with functional outcome by applying Spearman's rho and multivariate regression analysis. RESULTS: In all, 31/32 patients survived PML. A poor functional outcome was associated with higher age, higher initial John Cunningham virus (JCV) copy number in cerebrospinal fluid (CSF), and more extensive PML lesions on initial magnetic resonance imaging (MRI). No association between functional outcome and the duration of natalizumab therapy or a delayed PML diagnosis was observed. CONCLUSION: This study will be useful for neurological practice to estimate functional outcome or disease severity of natalizumab-PML in primary care settings.
OBJECTIVE: Progressive multifocal leukoencephalopathy (PML) is an emerging complication of immunosuppressive therapies, especially natalizumab in multiple sclerosis (MS). Factors associated with functional outcome of natalizumab-associated PML (natalizumab-PML) have not been sufficiently described. METHODS: We retrospectively analyzed medical records of all patients with natalizumab-PML ( n = 32) treated in our hospital since 2009. Disability measured by Expanded Disability Status Scale (EDSS) at two different time points (highest available EDSS during PML and last available EDSS after PML diagnosis) served as functional outcome parameters. Clinical, laboratory, and imaging data were analyzed for association with functional outcome by applying Spearman's rho and multivariate regression analysis. RESULTS: In all, 31/32 patients survived PML. A poor functional outcome was associated with higher age, higher initial John Cunningham virus (JCV) copy number in cerebrospinal fluid (CSF), and more extensive PML lesions on initial magnetic resonance imaging (MRI). No association between functional outcome and the duration of natalizumab therapy or a delayed PML diagnosis was observed. CONCLUSION: This study will be useful for neurological practice to estimate functional outcome or disease severity of natalizumab-PML in primary care settings.
Entities:
Keywords:
Expanded Disability Status Scale; John Cunningham Virus; Tysabri; long-term follow-up; multiple sclerosis; very late antigen-4
Authors: Martijn T Wijburg; Iris Kleerekooper; Birgit I Lissenberg-Witte; Marlieke de Vos; Clemens Warnke; Bernard M J Uitdehaag; Frederik Barkhof; Joep Killestein; Mike P Wattjes Journal: JAMA Neurol Date: 2018-07-01 Impact factor: 18.302
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