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https://www.readbyqxmd.com/read/29754703/progression-of-a-series-of-patients-with-relapsing-remitting-multiple-sclerosis-treated-for-7-years-with-natalizumab-using-the-no-evidence-of-disease-activity-parameter
#1
A Pato Pato, E Costa Arpín, A Rodríguez Regal, I Rodríguez Constenla, I Cimas Hernando, I Muñoz Pousa, L Naya Ríos, J R Lorenzo González, M C Amigo Jorrín, J M Prieto González
INTRODUCTION: The safety and effectiveness of natalizumab in patients with relapsing-remitting multiple sclerosis (RRMS) has been demonstrated in clinical trials. However, due to the limitations of these trials, it is important to know how the condition behaves under long-term clinical practice conditions. OBJECTIVE: To determine the long-term effectiveness of natalizumab in patients with RRMS by means of annual evaluation of the "no evidence of disease activity" (NEDA) parameter, which includes number of relapses, disability (measured with the Expanded Disability Status Scale), and brain MRI parameters...
May 10, 2018: Neurología: Publicación Oficial de la Sociedad Española de Neurología
https://www.readbyqxmd.com/read/29724903/natural-history-of-gata2-deficiency-in-a-survey-of-79-french-and-belgian-patients
#2
Jean Donadieu, Marie Lamant, Claire Fieschi, Flore Sicre de Fontbrune, Aurélie Caye, Marie Ouachee, Blandine Beaupain, Jacinta Bustamante, Hélène A Poirel, Bertrand Isidor, Eric Van Den Neste, Antoine Neel, Stanislas Nimubona, Fabienne Toutain, Vincent Barlogis, Nicolas Schleinitz, Thierry Leblanc, Pierre Rohrlich, Felipe Suarez, Dana Ranta, Wahid Abou Chahla, Bénédicte Bruno, Louis Terriou, Sylvie Francois, Bruno Lioure, Guido Ahle, Françoise Bachelerie, Claude Preudhomme, Eric Delabesse, Hélène Cavé, Christine Bellanné-Chantelot, Marlène Pasquet
Heterozygous germline GATA2 mutations strongly predispose to leukaemia, immunodeficiency, and/or lymphoedema. We now describe a series of 79 patients (53 families) diagnosed since 2011, compiling all patients in France and Belgium, with a follow up of 2249 patients/years. Median age at first clinical symptoms was 18.6 years (range, 0-61 years). Severe infectious diseases (mycobacteria, fungus, and human papilloma virus) and haematological malignancies were the most common first manifestations. The probability of remaining symptom-free was 8% at 40 years old...
May 3, 2018: Haematologica
https://www.readbyqxmd.com/read/29682547/jc-virus-dna-detection-is-associated-with-cd8-effector-accumulation-in-peripheral-blood-of-patients-with-multiple-sclerosis-under-natalizumab-treatment-independently-from-jc-virus-serostatus
#3
Maria A Zingaropoli, Marco Iannetta, Simona Pontecorvo, Elena Anzivino, Carla Prezioso, Donatella Maria Rodio, Manuela Morreale, Alessandra D'Abramo, Alessandra Oliva, Miriam Lichtner, Antonio Cortese, Marco Frontoni, Valeria Pietropaolo, Ada Francia, Claudio M Mastroianni, Vincenzo Vullo, Maria R Ciardi
Although natalizumab (anti- α 4 integrin) represents an effective therapy for relapsing remitting multiple sclerosis (RRMS), it is associated with an increased risk of developing progressive multifocal leukoencephalopathy (PML), caused by the polyomavirus JC (JCV). The aim of this study was to explore natalizumab-induced phenotypic changes in peripheral blood T-lymphocytes and their relationship with JCV reactivation. Forty-four patients affected by RRMS were enrolled. Blood and urine samples were classified according to natalizumab infusion number: 0 ( N 0), 1-12 ( N 12), 13-24 ( N 24), 25-36 ( N 36), and over 36 ( N > 36) infusions...
2018: BioMed Research International
https://www.readbyqxmd.com/read/29661457/progressive-multifocal-leukoencephalopathy-following-combined-rituximab-based-immune-chemotherapy-for-post-transplant-lymphoproliferative-disorder-in-a-renal-transplant-recipient-a-case-report
#4
M Windpessl, S Burgstaller, A Kronbichler, H Pieringer, O Kalev, A Karrer, M Wallner, J Thaler
BACKGROUND: Transplant recipients are at risk of developing progressive multifocal leukoencephalopathy (PML), an opportunistic infection due to reactivation of JC virus. Post-transplant lymphoproliferative disorders (PTLDs) represent a common malignancy in this population, and antiCD20-therapy has become an established component of its treatment. CASE PRESENTATION: We describe the first case of a renal allograft transplant recipient with PTLD who received rituximab-based immune-chemotherapy and developed PML shortly thereafter...
April 2018: Transplantation Proceedings
https://www.readbyqxmd.com/read/29656742/pathogenesis-of-progressive-multifocal-leukoencephalopathy-and-risks-associated-with-treatments-for-multiple-sclerosis-a-decade-of-lessons-learned
#5
REVIEW
Eugene O Major, Tarek A Yousry, David B Clifford
Progressive multifocal leukoencephalopathy (PML) is a rare, devastating demyelinating disease of the CNS caused by the JC virus (JCV) that occurs in patients with compromised immune systems. Detection of PML in systemically immunocompetent patients with multiple sclerosis treated with natalizumab points to a role for this drug in the pathophysiology of PML. Emerging knowledge of the cellular and molecular biology of JCV infection and the pathogenesis of PML-including interplay of this common virus with the human immune system and features of natalizumab that might contribute to PML pathogenesis-provides new opportunities to monitor viral status and predict risk of JCV-associated disease...
May 2018: Lancet Neurology
https://www.readbyqxmd.com/read/29532061/association-of-progressive-multifocal-leukoencephalopathy-lesion-volume-with-jc-virus-polymerase-chain-reaction-results-in-cerebrospinal-fluid-of-natalizumab-treated-patients-with-multiple-sclerosis
#6
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
Importance: The JC virus (JCV) was named after the first patient to be described with progressive multifocal leukoencephalopathy (PML), John Cunningham. Detection of JC virus DNA in cerebrospinal fluid (CSF) by polymerase chain reaction (PCR), and of specific lesions by brain magnetic resonance imaging (MRI), are both considered essential for the diagnosis of natalizumab-associated PML (NTZ-PML) in patients with multiple sclerosis. However, strict pharmacovigilance by MRI can result in detection of patients with small lesions and undetectable JCV DNA in CSF...
March 12, 2018: JAMA Neurology
https://www.readbyqxmd.com/read/29512846/progressive-multifocal-leukoencephalopathy-after-allogeneic-stem-cell-transplantation-case-report-and-review-of-the-literature
#7
Cai Yuan, Candace Deberardinis, Rushang Patel, Seema M Shroff, Steven A Messina, Steven Goldstein, Shahram Mori
Progressive multifocal leukoencephalopathy (PML) is a rare, yet typically fatal complication of allogeneic stem cell transplantation. It is caused by reactivation of the John Cunningham (JC) virus in an immunocompromised host. This report describes an unfortunate case of PML in a recipient of an allogeneic stem cell transplant for acute myelogenous leukemia. The JC virus was undetectable in the patient's cerebrospinal fluid by polymerase chain reaction (PCR); however, a positive diagnosis was made after a brain biopsy...
March 7, 2018: Transplant Infectious Disease: An Official Journal of the Transplantation Society
https://www.readbyqxmd.com/read/29472141/t-cell-deficiencies-as-a-common-risk-factor-for-drug-associated-progressive-multifocal-leukoencephalopathy
#8
REVIEW
Dejan Pavlovic, Mayur A Patel, Andriani C Patera, Ilse Peterson
Progressive multifocal leukoencephalopathy (PML) is a disease of the central nervous system caused by neuropathogenic prototypes of ubiquitous community-acquired JC virus (JCV). The disease became of particular concern following its association with certain therapies that modulate immune system function without heavy immunosuppression. Due to lack of prophylactic/treatment options and poor outcomes, which often include severe disability or death, PML is a considerable concern for development of new drugs that interfere with immune system functions...
June 2018: Immunobiology
https://www.readbyqxmd.com/read/29423619/smoking-is-not-associated-with-higher-prevalence-of-jc-virus-in-ms-patients
#9
Michael Auer, Gabriel Bsteh, Harald Hegen, Franziska Di Pauli, Sebastian Wurth, Thomas Berger, Florian Deisenhammer
John Cunningham virus (JCV) causes rare, but potentially life-threatening progressive multifocal leukoencephalopathy (PML) in natalizumab-treated multiple sclerosis (MS) patients. Beside JCV index, there is currently no other factor for further risk stratification. Because smoking was reported as potential risk factor for several viral and bacterial infections, we aimed to investigate whether smoking could increase the risk for JCV infection in MS patients. We screened our database of the MS Clinic of the Department of Neurology, Medical University of Innsbruck, Austria, for patients with known smoking status and test result for anti-JCV antibody index as determined by two-step ELISA at Unilabs, Copenhagen, Denmark...
May 2018: European Journal of Clinical Microbiology & Infectious Diseases
https://www.readbyqxmd.com/read/29414286/a-longitudinal-study-of-jc-virus-serostatus-stability-among-multiple-sclerosis-patients
#10
Raed Alroughani, Saeed Akhtar, Samar Ahmed, Jasem Al-Hashel
BACKGROUND: Anti-JC virus (JCV) antibody testing has been used increasingly to stratify multiple sclerosis (MS) patients into different disease modifying therapies. OBJECTIVES: We assessed JCV serostatus stability longitudinally in MS patients who were regularly tested for anti-JCV antibody and examined the demographic and clinical factors associated with JCV serostatus. METHODS: The data of MS patients who were screened for anti-JCV antibody using the two-step second-generation ELISA were collected between 1st October 2014 and 31st October 2015 at the MS clinic, Kuwait...
February 2018: Multiple Sclerosis and related Disorders
https://www.readbyqxmd.com/read/29396437/gene-therapy-for-human-glioblastoma-using-neurotropic-jc-virus-like-particles-as-a-gene-delivery-vector
#11
Chun-Nun Chao, Yu-Hsuan Yang, Mu-Sheng Wu, Ming-Chieh Chou, Chiung-Yao Fang, Mien-Chun Lin, Chien-Kuo Tai, Cheng-Huang Shen, Pei-Lain Chen, Deching Chang, Meilin Wang
Glioblastoma multiforme (GBM), the most common malignant brain tumor, has a short period of survival even with recent multimodality treatment. The neurotropic JC polyomavirus (JCPyV) infects glial cells and oligodendrocytes and causes fatal progressive multifocal leukoencephalopathy in patients with AIDS. In this study, a possible gene therapy strategy for GBM using JCPyV virus-like particles (VLPs) as a gene delivery vector was investigated. We found that JCPyV VLPs were able to deliver the GFP reporter gene into tumor cells (U87-MG) for expression...
February 2, 2018: Scientific Reports
https://www.readbyqxmd.com/read/29370683/systematic-review-of-the-published-data-on-the-worldwide-prevalence-of-john-cunningham-virus-in-patients-with-multiple-sclerosis-and-neuromyelitis-optica
#12
REVIEW
Sonia Patricia Castedo Paz, Luciana Branco, Marina Alves de Camargo Pereira, Caroline Spessotto, Yara Dadalti Fragoso
OBJECTIVES: John Cunningham virus (JCV) is a polyoma virus that infects humans, mainly in childhood or adolescence, and presents no symptomatic manifestations. JCV can cause progressive multifocal leukoencephalopathy (PML) in immunosuppressed individuals, including those undergoing treatment for multiple sclerosis (MS) and neuromyelitis optica (NMO). PML is a severe and potentially fatal disease of the brain. The prevalence of JCV antibodies in human serum has been reported to be between 50...
2018: Epidemiology and Health
https://www.readbyqxmd.com/read/29356206/survey-of-diagnostic-and-treatment-practices-for-multiple-sclerosis-ms-in-europe-part-2-progressive-ms-paediatric-ms-pregnancy-and-general-management
#13
O Fernández, M Delvecchio, G Edan, S Fredrikson, G Giovannoni, H-P Hartung, E Havrdova, L Kappos, C Pozzilli, P S Soerensen, B Tackenberg, P Vermersch, G Comi
BACKGROUND AND PURPOSE: The European Charcot Foundation supported the development of a set of surveys to understand current practice patterns for the diagnosis and management of multiple sclerosis (MS) in Europe. Part 2 of the report summarizes survey results related to secondary progressive MS (SPMS), primary progressive MS (PPMS), pregnancy, paediatric MS and overall patient management. METHODS: A steering committee of MS neurologists developed case- and practice-based questions for two sequential surveys distributed to MS neurologists throughout Europe...
May 2018: European Journal of Neurology: the Official Journal of the European Federation of Neurological Societies
https://www.readbyqxmd.com/read/29346632/diagnostic-and-prognostic-value-of-jc-virus-dna-in-plasma-in-progressive-multifocal-leukoencephalopathy
#14
Francesca Ferretti, Arabella Bestetti, Constantin T Yiannoutsos, Beverly S Musick, Simonetta Gerevini, Laura Passeri, Simona Bossolasco, Antonio Boschini, Diego Franciotta, Adriano Lazzarin, Paola Cinque
Background: Progressive multifocal leukoencephalopathy (PML) is a severe demyelinating disease caused by the polyomavirus JC (JCV) affecting subjects with impaired immune system. While JCV-DNA detection in cerebrospinal fluid (CSF) is diagnostic of PML, the clinical significance of plasma JCV-DNA is still uncertain. Methods: We retrospectively analyzed plasma samples drawn from patients with PML close to disease onset, and controls without PML. In PML patients, we compared plasma JCV DNA detection and levels to: JCV DNA in the other biological samples, clinical and laboratory parameters and patients' survival...
January 15, 2018: Clinical Infectious Diseases: An Official Publication of the Infectious Diseases Society of America
https://www.readbyqxmd.com/read/29321332/erk-is-a-critical-regulator-of-jc-polyomavirus-infection
#15
Jeanne K DuShane, Michael P Wilczek, Colleen L Mayberry, Melissa S Maginnis
The human JC polyomavirus (JCPyV) infects the majority of the population worldwide and presents as an asymptomatic, persistent infection in the kidneys. In individuals who are immunocompromised, JCPyV can become reactivated and cause a lytic infection in the central nervous system resulting in the fatal, demyelinating disease progressive multifocal leukoencephalopathy (PML). Infection is initiated by interactions between the capsid protein viral protein 1 (VP1) and the α2,6-linked sialic acid on lactoseries tetrasaccharide c (LSTc), while JCPyV internalization is facilitated by 5-hydroxytryptamine 2 receptors (5-HT2 Rs)...
April 1, 2018: Journal of Virology
https://www.readbyqxmd.com/read/29159704/neuroimmune-regulation-of-jc-virus-by-intracellular-and-extracellular-agnoprotein
#16
Michael Craigie, Stephanie Cicalese, Ilker Kudret Sariyer
JC virus (JCV) is a human polyomavirus and the etiologic agent of the demyelinating disease progressive multifocal leukoencephalopathy (PML). PML is observed in patients with underlying immunocompromising conditions, suggesting that neuro-immune interactions between peripheral immune cells and neuro-glia play an important role in controlling viral reactivation in the brain. There is little known about the immunobiology of JCV reactivation in glial cells and the role of immune, glial, and viral players in this regulation...
November 20, 2017: Journal of Neuroimmune Pharmacology: the Official Journal of the Society on NeuroImmune Pharmacology
https://www.readbyqxmd.com/read/29139004/progressive-multifocal-leukoencephalopathy-in-the-absence-of-immunosuppression
#17
Benjamin E Zucker, Sybil R L Stacpoole
A 69-year-old woman presented with a cortical hand syndrome progressing over several weeks. MRI brain showed characteristic appearances of progressive multifocal leukoencephalopathy (PML), confirmed by detection of the JC virus in CSF, despite the absence of any evidence of immunosuppression. Treatment with mirtazapine, mefloquine and cidofovir did not affect the progression of the disease, which was fatal within 7 months of presentation. This report adds to the small case literature that suggests that PML can occur in immunocompetent people, albeit extremely rarely...
February 2018: Journal of Neurovirology
https://www.readbyqxmd.com/read/29120025/prevention-and-therapy-of-jc-polyomavirus-mediated-progressive-multifocal-leukoencephalopathy-a-realistic-possibility
#18
Ivan Jelcic, Benoit Combaluzier, Ilijas Jelcic, Mireia Sospedra, Jan Grimm, Roland Martin
Progressive multifocal leukoencephalopathy (PML) is a serious opportunistic infection of the brain caused by the JC polyomavirus (JCPyV). PML occurs when immune control of persistent infection with JCPyV fails, the virus mutates and changes its cellular tropism, enters the brain and infects astrocytes, oligodendrocytes and, in particular cases, also neurones. Currently, there is no therapy for this often fatal disease. A number of approaches have failed, and only the restoration of protective immunity, if possible, can lead to clearance of the virus once PML has occurred...
November 9, 2017: Swiss Medical Weekly
https://www.readbyqxmd.com/read/29098656/virological-quality-of-irrigation-water-sources-and-pepper-mild-mottle-virus-and-tobacco-mosaic-virus-as-index-of-pathogenic-virus-contamination-level
#19
Sadhana Shrestha, Shankar Shrestha, Junko Shindo, Jeevan B Sherchand, Eiji Haramoto
Irrigation water is a doorway for the pathogen contamination of fresh produce. We quantified pathogenic viruses [human adenoviruses, noroviruses of genogroups I and II, group A rotaviruses, Aichi virus 1 (AiV-1), enteroviruses (EnVs), and salivirus (SaliV)] and examined potential index viruses [JC and BK polyomaviruses (JCPyVs and BKPyVs), pepper mild mottle virus (PMMoV), and tobacco mosaic virus (TMV)] in irrigation water sources in the Kathmandu Valley, Nepal. River, sewage, wastewater treatment plant (WWTP) effluent, pond, canal, and groundwater samples were collected in September 2014, and in April and August 2015...
March 2018: Food and Environmental Virology
https://www.readbyqxmd.com/read/29068484/the-clinical-utility-of-jc-virus-antibody-index-measurements-in-the-context-of-progressive-multifocal-leukoencephalopathy
#20
REVIEW
A Q Reuwer, M Heron, D van der Dussen, T Schneider-Hohendorf, J L Murk
In natalizumab-treated patients without previous immunosuppressive treatment, the JCV antibody index is used to stratify PML risk. A high index value indicates that the risk to develop PML is significantly elevated, although probably about 99% of patients with this index value will not develop PML. This minireview aimed to provide an overview of the basic virology and immunology relevant to understanding JCV infections in MS patients, with a focus on what is presently known about antibodies to JCV and how they could be of use to predict and diagnose PML...
November 2017: Acta Neurologica Scandinavica
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