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https://www.readbyqxmd.com/read/28099331/atorvastatin-induced-necrotizing-autoimmune-myositis-an-emerging-dominant-entity-in-patients-with-autoimmune-myositis-presenting-with-a-pure-polymyositis-phenotype
#1
Yves Troyanov, Océane Landon-Cardinal, Marvin J Fritzler, José Ferreira, Ira N Targoff, Eric Rich, Michelle Goulet, Jean-Richard Goulet, Josiane Bourré-Tessier, Yves Robitaille, Julie Drouin, Alexandra Albert, Jean-Luc Senécal
The general aim of this study was to evaluate the disease spectrum in patients presenting with a pure polymyositis (pPM) phenotype. Specific objectives were to characterize clinical features, autoantibodies (aAbs), and membrane attack complex (MAC) in muscle biopsies of patients with treatment-responsive, statin-exposed necrotizing autoimmune myositis (NAM). Patients from the Centre hospitalier de l'Université de Montréal autoimmune myositis (AIM) Cohort with a pPM phenotype, response to immunosuppression, and follow-up ≥3 years were included...
January 2017: Medicine (Baltimore)
https://www.readbyqxmd.com/read/28097746/a-high-neutrophil-to-lymphocyte-ratio-is-associated-with-refractory-kawasaki-disease
#2
Hong-Je Cho, So Young Bak, Su Yeong Kim, Rita Yoo, Hae-Sung Baek, Seung Yang, Il-Tae Hwang, Ji-Eun Ban
BACKGROUND: The clinical significance of the neutrophil to lymphocyte ratio (NLR) has not yet been fully elucidated in patients with Kawasaki disease (KD). The purpose of this study is to investigate the relationship between NLR and the response to intravenous immunoglobulin (IVIG) therapy affecting coronary abnormalities in KD METHODS: A total of 196 patients of KD who were treated with IVIG therapy were analyzed. We evaluated the baseline NLR immediately before IVIG therapy and classified the study patients into 2 groups according to the value of the NLR...
January 18, 2017: Pediatrics International: Official Journal of the Japan Pediatric Society
https://www.readbyqxmd.com/read/28095967/intravenous-immunoglobulin-therapy-in-pediatric-narcolepsy-a-nonrandomized-open-label-controlled-longitudinal-observational-study
#3
Michel Lecendreux, Johanna Berthier, Jennifer Corny, Olivier Bourdon, Claire Dossier, Christophe Delclaux
STUDY OBJECTIVES: Previous case reports of intravenous immunoglobulins (IVIg) in pediatric narcolepsy have shown contradictory results. METHODS: This was a nonrandomized, open-label, controlled, longitudinal observational study of IVIg use in pediatric narcolepsy with retrospective data collection from medical files obtained from a single pediatric national reference center for the treatment of narcolepsy in France. Of 56 consecutively referred patients with narcolepsy, 24 received IVIg (3 infusions administered at 1-mo intervals) in addition to standard care (psychostimulants and/or anticataplectic agents), and 32 continued on standard care alone (controls)...
January 11, 2017: Journal of Clinical Sleep Medicine: JCSM: Official Publication of the American Academy of Sleep Medicine
https://www.readbyqxmd.com/read/28093780/subacute-demyelinating-polyradiculoneuropathy-complicating-epstein-barr-virus-infection-in-gata2-haploinsufficiency
#4
Mohamed Kazamel, Christopher J Klein, Eduardo E Benarroch, Mrinal M Patnaik, Jennifer A Tracy
INTRODUCTION: Autosomal dominant haploinsufficiency of GATA2 causes monocytopenia and natural killer cell lymphopenia, resulting in predisposition to mycobacterial, fungal, and viral infections. METHODS: We report clinical, serologic, electrophysiologic, and pathologic evaluation of a 29-year-old woman with GATA2 haploinsufficiency and active Epstein-Barr virus (EBV) infection complicated by subacute painful neuropathy. RESULTS: Nerve conduction and electromyography studies showed predominantly demyelinating sensorimotor polyradiculoneuropathy...
January 17, 2017: Muscle & Nerve
https://www.readbyqxmd.com/read/28092085/evidence-that-nf-%C3%AE%C2%BAb-and-mapk-signaling-promotes-nlrp-inflammasome-activation-in-neurons-following-ischemic-stroke
#5
David Yang-Wei Fann, Yun-An Lim, Yi-Lin Cheng, Ker-Zhing Lok, Prasad Chunduri, Sang-Ha Baik, Grant R Drummond, S Thameem Dheen, Christopher G Sobey, Dong-Gyu Jo, Christopher Li-Hsian Chen, Thiruma V Arumugam
Multi-protein complexes, termed "inflammasomes," are known to contribute to neuronal cell death and brain injury following ischemic stroke. Ischemic stroke increases the expression and activation of nucleotide-binding oligomerization domain (NOD)-like receptor (NLR) Pyrin domain containing 1 and 3 (NLRP1 and NLRP3) inflammasome proteins and both interleukin (IL)-1β and IL-18 in neurons. In this study, we provide evidence that activation of either the NF-κB and MAPK signaling pathways was partly responsible for inducing the expression and activation of NLRP1 and NLRP3 inflammasome proteins and that these effects can be attenuated using pharmacological inhibitors of these two pathways in neurons and brain tissue under in vitro and in vivo ischemic conditions, respectively...
January 14, 2017: Molecular Neurobiology
https://www.readbyqxmd.com/read/28091723/changes-in-the-severity-and-subtype-of-guillain-barr%C3%A3-syndrome-admitted-to-a-specialist-neuromedical-icu-over-a-25%C3%A2-year-period
#6
C J Taylor, N P Hirsch, D M Kullmann, Robin S Howard
We report a retrospective review of 110 patients with acute Guillain-Barré syndrome (GBS) admitted to a specialised intensive care unit (ICU) in a tertiary referral centre over a 25 year period, the start of which coincided with the widespread introduction of plasma exchange (PE) and intravenous immunoglobulin (IVIG). The results were analysed by comparing 52 patients admitted in the first decade (1991-2000; Group 1) with 58 patients admitted between 2001-2014 (Group 2). Patients in both groups were comparable with respect to age and sex, and had a similar incidence and range of ICU complications...
January 16, 2017: Journal of Neurology
https://www.readbyqxmd.com/read/28089146/factors-associated-with-development-of-coronary-artery-aneurysms-after-kawasaki-disease-are-similar-for-those-treated-promptly-and-those-with-delayed-or-no-treatment
#7
Mallory L Downie, Cedric Manlhiot, Tanveer H Collins, Nita Chahal, Rae S M Yeung, Brian W McCrindle
BACKGROUND: While the risk is reduced, patients may develop coronary artery (CA) aneurysms after Kawasaki disease (KD) despite receiving intravenous immunoglobulin (IVIG) within 10days of symptom onset. Risk factors for CA aneurysms may differ compared to those patients with delayed or no treatment. METHODS: Patients diagnosed with KD between 1990 and 2013 were included. Patients with maximum coronary artery z-scores>5 were classified as having CA aneurysms. Separate multivariable regression models were used to determine factors associated with CA aneurysms for those with versus without prompt treatment...
January 8, 2017: International Journal of Cardiology
https://www.readbyqxmd.com/read/28087884/the-role-of-immunoglobulin-prophylaxis-for-prevention-of-cytomegalovirus-infection-in-pediatric-hematopoietic-stem-cell-transplantation-recipients
#8
Gal Goldstein, Tal Frenkel Rutenberg, Sarina Levy Mendelovich, Daphna Hutt, Michal Teperberg Oikawa, Amos Toren, Bella Bielorai
BACKGROUND: Following cessation of intravenous immunoglobulin (IVIg) administration for allogeneic hematopoietic stem cell transplantation (HSCT) recipients at our unit, we observed a sharp decline in the incidence of cytomegalovirus (CMV) infection. PROCEDURE: We conducted a retrospective study of the role of IVIg in the prevention of CMV infection in children and young adults who underwent HSCT from matched related donor. RESULTS: We included 109 patients (IVIg+/IVIg- ratio 82/27)...
January 14, 2017: Pediatric Blood & Cancer
https://www.readbyqxmd.com/read/28079915/intravenous-immunoglobulin-for-the-treatment-of-kawasaki-disease
#9
Stanford T Shulman
Standard first-line therapy for Kawasaki disease (KD) consists of intravenous immunoglobulin (IVIG) and aspirin. Current guidelines recommend 2 g/kg of IVIG and 80 to 100 mg/kg of aspirin administered within the first 10 days of illness. This regimen has marked efficacy in preventing the development of coronary artery aneurysms. Approximately 15% to 20% of treated patients require a second dose of IVIG to control the inflammatory process. The role of adjunctive corticosteroid therapy with IVIG and aspirin is evolving, with Japanese studies showing a clear benefit in those patients at highest risk for development of coronary disease...
January 1, 2017: Pediatric Annals
https://www.readbyqxmd.com/read/28079914/intravenous-immunoglobulin-in-pediatric-rheumatology-when-to-use-it-and-what-is-the-evidence
#10
Martha M Rodriguez, Linda Wagner-Weiner
Intravenous immunoglobulin (IVIG) is given to children with a variety of rheumatologic illnesses. The mechanism of action by which it exerts therapeutic effects is not well understood and likely differs in the medical conditions for which it is given. IVIG is approved by the US Food and Drug Administration and is the standard of care for Kawasaki disease, but most IVIG use in pediatric rheumatology is "off-label. " The literature supports the use of IVIG for juvenile dermatomyositis, although it is unclear whether its use should be limited to those children with more severe or refractory disease...
January 1, 2017: Pediatric Annals
https://www.readbyqxmd.com/read/28079913/intravenous-immunoglobulin-in-the-treatment-of-hematologic-disorders-in-pediatrics
#11
Gabriela Villanueva, Jill L O de Jong, Jennifer L McNeer
Intravenous immunoglobulin (IVIG) is pooled immunoglobulin G derived from human blood donors. It was introduced in the early 1980s to treat immunodeficiency disorders. Since then, its use has expanded to other fields such as neurology, rheumatology, and hematology. IVIG has been used to provide passive immunity in qualitative and quantitative immunoglobulin disorders, to neutralize antibodies in immune-mediated diseases, and as an immune modulatory agent. The difficulty of producing IVIG in high quantities, in addition to a growing list of "off-label" indications, has resulted in a worldwide shortage and increase in cost...
January 1, 2017: Pediatric Annals
https://www.readbyqxmd.com/read/28079912/intravenous-immunoglobulin-in-the-treatment-of-primary-immunodeficiency-diseases
#12
Deirdre De Ranieri, Nana Sarkoah Fenny
Intravenous immunoglobulin (IVIG) has been used as antibody replacement therapy in primary immunodeficiency diseases (PIDDs) for more than 50 years. Its role as a therapeutic agent has expanded over the past couple of decades as its anti-inflammatory and immune-modulatory mechanisms of action have been elucidated. It is now used "off-label" to treat other autoimmune diseases. This article focuses on the role of IVIG in the treatment of PIDDs characterized by absent or deficient antibody production. Replacement doses are given on a monthly basis in these conditions as a prophylactic measure to prevent acute and serious bacterial infections...
January 1, 2017: Pediatric Annals
https://www.readbyqxmd.com/read/28079804/aquaporin-4-positive-neuromyelitis-optica-spectrum-disorders-secondary-to-thrombopenic-purpura-a-case-report
#13
Ying Wang, Qiaoyun Gong, Mingqin Zhu, Chao Lu, Li Sun, Jiachun Feng, Hongliang Zhang
RATIONALE: Neuromyelitis optica spectrum disorders (NMOSD) is considered as an immune-mediated disorder in the central nervous system (CNS). Numerous autoimmune diseases are frequently complicated with NMOSD and distinct clinical characteristics are noted in NMOSD patients with other autoimmune diseases. However, to our best knowledge, co-occurrence of NMOSD and thrombopenic purpura is rarely identified. PATIENT CONCERNS: We presented a rare case of a 72-year-old female with 6-year history of thrombopenic purpura, and 1-month history of blurred vision as well as chest zonethesia...
January 2017: Medicine (Baltimore)
https://www.readbyqxmd.com/read/28079513/high-dose-aspirin-for-kawasaki-disease-outdated-myth-or-effective-aid
#14
Gil Amarilyo, Yael Koren, Dafna Brik Simon, Maskit Bar-Meir, Hilla Bahat, Mona Hanna Helou, Amir Mendelson, Nofar Hezkelo, Gabriel Chodick, Yackov Berkun, Eli Eisenstein, Yonatan Butbul Aviel, Galia Barkai, Yoav Bolkier, Shai Padeh, Riva Brik, Phillip J Hashkes, Liora Harel, Yosef Uziel
OBJECTIVES: To compare the efficacy and safety of intravenous immunoglobulin (IVIG) plus high-dose aspirin (HDA) vs. IVIG plus low-dose aspirin (LDA) for the treatment of Kawasaki disease, with an emphasis on coronary artery outcomes. METHODS: This study was a retrospective, medical record review of paediatric patients with Kawasaki disease comparing 6 centres that routinely used HDA for initial treatment and 2 that used LDA in 2004-2013. Treatment response and adverse events were compared...
January 5, 2017: Clinical and Experimental Rheumatology
https://www.readbyqxmd.com/read/28076913/use-of-intravenous-immunoglobulin-therapy-for-myositis-an-audit-in-south-australian-patients
#15
Caroline Foreman, Paul Russo, Noelene Davies, Pravin Hissaria, Susanna Proudman, Tiffany Hughes, Vidya Limaye
In South Australia, between 2000 and 2014, 57 patients with idiopathic inflammatory myositis (IIM) were treated with intravenous immunoglobulin (IVIg). We reviewed disease characteristics to determine predictors of response to therapy and IVIg dosing and duration to identify opportunities to rationalise IVIg use. Patients with dermatomyositis/polymyositis had a response rate of 77% and were more likely than inclusion body myositis to respond to therapy. Consideration should be given to the use of the lowest possible dose of IVIg and to the undertaking of trials of cessation of IVIg in patients with stable IIM...
January 2017: Internal Medicine Journal
https://www.readbyqxmd.com/read/28074934/disease-modifying-effect-of-intravenous-immunoglobulin-in-an-experimental-model-of-epilepsy
#16
Min Chen, Thiruma V Arumugam, Gayeshika Leanage, Quang M Tieng, Ashwin Yadav, Jeremy F P Ullmann, David T She, Vy Truong, Marc J Ruitenberg, David C Reutens
Novel therapies that prevent or modify the development of epilepsy following an initiating brain insult could significantly reduce the burden of this disease. In light of evidence that immune mechanisms play an important role in generating and maintaining the epileptic condition, we evaluated the effect of a well-established immunomodulatory treatment, intravenous immunoglobulin (IVIg), on the development of epilepsy in an experimental model of epileptogenesis. In separate experiments, IVIg was administered either before (pre-treatment) or after (post-treatment) the onset of pilocarpine status epilepticus (SE)...
January 11, 2017: Scientific Reports
https://www.readbyqxmd.com/read/28073042/rapid-infusions-of-human-normal-immunoglobulin-50g-l-are-safe-and-well-tolerated-in-immunodeficiencies-and-immune-thrombocytopenia
#17
Giuseppe Spadaro, Alessandra Vultaggio, A Alberto Bosi, Dietmar Reichert, Jan Janssen, Donatella Lamacchia, Liliana Nappi, Antonio Pecoraro, Cinzia Milito, Andrea Ferraro, Andrea Matucci, Francesca Bacchiarri, Valentina Carrai, Azra Hibbeler, Elisabet Speckman, Chiara Guarnieri, Serena Bongiovanni, Isabella Quinti
Intravenous immunoglobulin (IVIg) is accepted as an effective and well-tolerated treatment for primary and secondary immunodeficiencies (ID) and immune thrombocytopenia (ITP). Adverse reactions of IVIg are usually mild, comprising transient flu-like symptoms, change in blood pressure and tachycardia. However IVIg therapy can be burdensome for both patients and healthcare facilities, since the infusion may take up to 4h to administer. The objective of our multicentre, prospective, open-label phase III trial was to evaluate the tolerability and safety of human normal immunoglobulin 50g/l (Ig VENA) at high intravenous infusion rates in adult patients with ID and ITP who had previously tolerated IVIg treatment, by progressively increasing infusion rate up to 8ml/kg/hr...
January 7, 2017: International Immunopharmacology
https://www.readbyqxmd.com/read/28072954/-clinical-and-immunological-analysis-of-patients-with-activated-phosphoinositide-3-kinase-%C3%AE-syndrome-resulting-from-pik3cd-mutation
#18
W J Tang, W Wang, Y Luo, Y P Wang, L Li, Y F An, L J Gou, M S Ma, T Y He, J Yang, X D Zhao, H M Song
Objective: To explore the clinical and immunological features, gene mutations, treatment and prognosis in patients with activated phosphoinositide 3-kinase δ syndrome (APDS) caused by PIK3CD gene heterozygous germline mutation. Method: The data of clinical, immunological phenotype, treatment, and prognosis of 15 patients with APDS, who visited Children's Hospital of Chongqing Medical University, Peking Union Medical College Hospital, and Shenzhen Children's Hospital from June 2014 to November 2016, were collected and analyzed...
January 2, 2017: Zhonghua Er Ke za Zhi. Chinese Journal of Pediatrics
https://www.readbyqxmd.com/read/28069066/anti-inflammatory-effect-of-resveratrol-in-human-coronary-arterial-endothelial-cells-via-induction-of-autophagy-implication-for-the-treatment-of-kawasaki-disease
#19
Fu-Chen Huang, Ho-Chang Kuo, Ying-Hsien Huang, Hong-Ren Yu, Sung-Chou Li, Hsing-Chun Kuo
BACKGROUND: Kawasaki disease (KD) is an acute febrile vasculitis in childhood, which is the leading cause of acquired heart disease in children. If untreated, KD can result in coronary aneurysms in 25% of patients, and even under intravenous immunoglobulin (IVIG) treatment, 10-20% of children will have IVIG resistance and increased risk of developing coronary arteritis complication. Additional therapies should be explored to decrease the incidence of coronary artery lesions and improve the prognosis in KD...
January 9, 2017: BMC Pharmacology & Toxicology
https://www.readbyqxmd.com/read/28068584/intravenous-immunoglobulins-for-refractory-status-epilepticus-part-i-a-scoping-systematic-review-of-the-adult-literature
#20
REVIEW
F A Zeiler, M Matuszczak, J Teitelbaum, C J Kazina, L M Gillman
PURPOSE: Our goal was to perform a scoping systematic review of the literature on the use of intravenous immunoglobulins (IVIG) for refractory status epilepticus (RSE) in adults. METHOD: Articles from MEDLINE, BIOSIS, EMBASE, Global Health, Healthstar, Scopus, Cochrane Library, the International Clinical Trials Registry Platform, clinicaltrials.gov (inception to May 2016), reference lists of relevant articles, and gray literature were searched. The strength of evidence was adjudicated using both the Oxford and GRADE methodology by two independent reviewers...
December 27, 2016: Seizure: the Journal of the British Epilepsy Association
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