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https://www.readbyqxmd.com/read/28444889/invasive-fungal-infections-in-paediatric-patients-treated-with-macromolecular-immunomodulators-other-than-tumour-necrosis-alpha-inhibitors
#1
REVIEW
Ioannis Kyriakidis, Athanasios Tragiannidis, Ilse Zündorf, Andreas H Groll
An expanding list of immunomodulatory or immunosuppressive monoclonal antibodies (mAbs) and biologic therapeutics is currently entering clinical practice, particularly in the areas of oncology, transplantation and autoimmune disorders. These agents are directed against molecules or cells involved in inflammation and immunity and may therefore be associated with serious and opportunistic infections. The purpose of this review was to critically analyse the literature on invasive fungal infections (IFIs) occurring in association with mAbs and fusion proteins other than tumour necrosis alpha (TNF-α) inhibitors, including therapeutics modulating T-cell-mediated pathologies (muromonab, abatacept, belatacept, ipilimumab, basiliximab, daclizumab), inducing lymphopenia (alemtuzumab), depleting CD20+ B cells (rituximab) and interfering with various targets (anakinra, natalizumab, blodalumab, ixekizumab and others) with a focus on children, and to provide a framework of evaluating the risk for IFIs in this population...
April 26, 2017: Mycoses
https://www.readbyqxmd.com/read/28427379/novel-mutation-identified-in-severe-early-onset-tumor-necrosis-factor-receptor-associated-periodic-syndrome-a-case-report
#2
Suhas M Radhakrishna, Amy Grimm, Lori Broderick
BACKGROUND: Tumor Necrosis Factor Receptor-Associated Periodic Syndrome (TRAPS) is the second most common heritable autoinflammatory disease, typically presenting in pre-school aged children with fever episodes lasting 1-3 weeks. Systemic symptoms can include rash, myalgia, ocular inflammation, and serositis. CASE PRESENTATION: Here we report an unusual presentation of TRAPS in a 7 month old girl who presented with only persistent fever. She was initially diagnosed with incomplete Kawasaki Disease and received IVIG and infliximab; however, her fevers quickly recurred...
April 20, 2017: BMC Pediatrics
https://www.readbyqxmd.com/read/28418334/juvenile-idiopathic-arthritis
#3
Kenan Barut, Amra Adrovic, Sezgin Şahin, Özgür Kasapçopur
Juvenile idiopathic arthritis is the most common chronic rheumatic disease of unknown aetiology in childhood and predominantly presents with peripheral arthritis. The disease is divided into several subgroups, according to demographic characteristics, clinical features, treatment modalities and disease prognosis. Systemic juvenile idiopathic arthritis, which is one of the most frequent disease subtypes, is characterized by recurrent fever and rash. Oligoarticular juvenile idiopathic arthritis, common among young female patients, is usually accompanied by anti-nuclear antibodie positivity and anterior uveitis...
April 5, 2017: Balkan Medical Journal
https://www.readbyqxmd.com/read/28416939/a-systemic-onset-juvenile-idiopathic-arthritis-patient-with-reduced-anakinra-treatment-admitted-with-an-attack
#4
Dilek Yilmaz, Mediha Akcan, Semiha Terlemez, Ferah Sonmez, Abdullah Baris Akcan
Interleukin-1 plays an important role in the pathogenesis of systemic-onset juvenile idiopathic arthritis (SoJIA), and the use of anti-interleukin-1 therapy has been increasing. We report a case of a 14-year-old male patient with SoJIA. He was in remission with anakinra treatment for almost 2 years. When we extended the therapeutic range and decreased the dose (1 mg/kg twice a week), he developed symptoms mimicking pulmonary embolism and cardiac ischemia. Increased cardiac enzyme levels and echocardiographic findings were interpreted as myopericarditis...
February 2017: Eurasian Journal of Medicine
https://www.readbyqxmd.com/read/28400731/il-1-inhibition-may-have-an-important-role-in-treating-refractory-kawasaki-disease
#5
REVIEW
Perrine Dusser, Isabelle Koné-Paut
Kawasaki disease (KD) is an acute inflammatory vasculitis occurring in young children before 5 years and representing at this age, the main cause of acquired heart disease. A single infusion of 2 g/kg of intravenous immunoglobulins along with aspirin has reduced the frequency of coronary artery aneurysms from 25 to 5%. However, 10-20% of patients do not respond to standard treatment and have an increased risk of cardiac complications and death. The development of more potent therapeutic approaches of KD is an urgent need...
2017: Frontiers in Pharmacology
https://www.readbyqxmd.com/read/28390409/a-child-with-resistant-kawasaki-disease-successfully-treated-with-anakinra-a-case-report
#6
J Sánchez-Manubens, A Gelman, N Franch, S Teodoro, J R Palacios, N Rudi, J Rivera, J Antón
BACKGROUND: Kawasaki disease (KD) is an acute self-limited systemic vasculitis of unknown etiology. Intravenous immunoglobulin (IVIG) is an effective treatment and decreases the risk of cardiac complications to less than 5%. In spite of its effectiveness, some children do not respond to this therapy and still develop coronary aneurysms (CAA). The optimal treatment for IVIG non-responsive patients remains controversial although corticoids have been suggested to be an effective treatment in some patients...
April 8, 2017: BMC Pediatrics
https://www.readbyqxmd.com/read/28371574/tofacitinib-suppresses-disease-activity-and-febrile-attacks-in-a-patient-with-coexisting-rheumatoid-arthritis-and-familial-mediterranean-fever
#7
Kevser Gök, Gizem Cengiz, Kemal Erol, Salih Ozgocmen
Familial Mediterranean fever (FMF) is the most common hereditary auto-inflammatory (periodic fever) syndrome, and usually successfully treated with colchicine. However, nearly 5-10% of FMF cases are resistant or intolerant to colchicine and treatment options are highly restricted in these cases. Biologics including anakinra, canakinumab, rilonacept, etanercept, infliximab, interferon-alpha, and tocilizumab are shown to have efficacy to control FMF attacks. Tofacitinib, a Janus kinase (JAK) inhibitor, is an orally administered non-biologic disease modifying anti-rheumatic drug for the treatment of rheumatoid arthritis (RA)...
January 2017: Acta Reumatológica Portuguesa
https://www.readbyqxmd.com/read/28367532/the-effect-of-anakinra-on-retinal-function-in-isolated-perfused-vertebrate-retina
#8
Mahdy Ranjbar, Toni Schneider, Carl Brand, Salvatore Grisanti, Julia Lüke, Matthias Lüke
PURPOSE: Blockage of the interleukin 1 (IL-1) signaling pathway has been proposed for treatment of inflammatory disorders like those affecting the retina and its adjacent tissue. Herein, we evaluated one of those inhibitory drugs, anakinra (Kineret(®)), based on its safety profile with emphasis on retinal function from an electrophysiological point of view. METHODS: Bovine retina preparations were perfused with two different concentrations of anakinra (1 mg/ml and 2 mg/ml)...
March 2017: Journal of Current Ophthalmology
https://www.readbyqxmd.com/read/28347403/lack-of-il-1r8-in-neurons-causes-hyperactivation-of-il-1-receptor-pathway-and-induces-mecp2-dependent-synaptic-defects
#9
Romana Tomasoni, Raffaella Morini, Jose P Lopez-Atalaya, Irene Corradini, Alice Canzi, Marco Rasile, Cristina Mantovani, Davide Pozzi, Cecilia Garlanda, Alberto Mantovani, Elisabetta Menna, Angel Barco, Michela Matteoli
Inflammation modifies risk and/or severity of a variety of brain diseases through still elusive molecular mechanisms. Here we show that hyperactivation of the interleukin 1 pathway, through either ablation of the interleukin 1 receptor 8 (IL-1R8, also known as SIGIRR or Tir8) or activation of IL-1R, leads to up-regulation of the mTOR pathway and increased levels of the epigenetic regulator MeCP2, bringing to disruption of dendritic spine morphology, synaptic plasticity and plasticity-related gene expression...
March 28, 2017: ELife
https://www.readbyqxmd.com/read/28337831/interleukin-1-alpha-deficiency-increases-the-expression-of-follicle-stimulating-hormone-receptors-in-granulosa-cells
#10
Shiri Uri-Belapolsky, Irit Miller, Aviv Shaish, Mattan Levi, Dror Harats, Lihi Ninio-Many, Yehuda Kamari, Ruth Shalgi
Follicle-stimulating hormone receptor (FSHR) is a pivotal regulator of ovarian response to hormonal stimulation. Inflammatory conditions have been linked to lower FSHR expression in granulosa cells (GCs) as well as an attenuated response to hormonal stimulation. The current study aimed to reveal if deficiency and/or blockage of the pro-inflammatory cytokine interleukin 1-alpha (IL1A) increased Fshr expression in rodent GCs. We found elevated Fshr transcript abundance, as assessed by quantitative PCR, in primary GCs isolated from Il1a-knockout compared to wild-type mice, and that the expression of FSHR is significantly higher in Il1a-knockout compared to wild-type ovaries...
March 24, 2017: Molecular Reproduction and Development
https://www.readbyqxmd.com/read/28335798/treatment-of-mucocutaneous-manifestations-in-beh%C3%A3-et-s-disease-with-anakinra-a-pilot-open-label-study
#11
Peter C Grayson, Yusuf Yazici, Melissa Merideth, H Nida Sen, Michael Davis, Elaine Novakovich, Elizabeth Joyal, Raphaela Goldbach-Mansky, Cailin H Sibley
BACKGROUND: The effect of IL-1 blocking therapy on mucocutaneous manifestations of Behçet's disease is incompletely understood. METHODS: Six patients with Behçet's disease and ongoing oral/genital ulcers for ≥1 month were enrolled into an adaptive, two-phase clinical trial and included in the analysis. Study duration was 6 months with extension up to 16 months. All were treated non-blinded with anakinra 100 mg subcutaneous daily with the option to escalate the dose to 200 mg in partial responders after 1 month and 300 mg after 6 months...
March 24, 2017: Arthritis Research & Therapy
https://www.readbyqxmd.com/read/28333874/successful-treatment-of-autoimmune-disease-associated-pyoderma-gangrenosum-with-the-il-1-receptor-antagonist-anakinra-a-case-series-of-3-patients
#12
Celia Beynon, Mei Fong Chin, Prasad Hunasehally, Krishnan Bhagwandas, Martin Bevan, Marisa Taylor, Thomas Lawson
No abstract text is available yet for this article.
April 2017: Journal of Clinical Rheumatology: Practical Reports on Rheumatic & Musculoskeletal Diseases
https://www.readbyqxmd.com/read/28326845/the-use-of-biologics-in-pregnant-patients-with-rheumatic-disease
#13
Monika Østensen
An increasing number of female patients with autoimmune diseases are treated with biologic drugs. Concerns in regard to safety of biologics during pregnancy arise in patients who have not completed their families. Areas covered: A review of the literature dealing with child outcomes of pregnancies exposed to biologics shows that TNF inhibitors (TNFi) are the best studied in regard to human pregnancy. In studies comparing exposed pregnancies to disease-matched controls no increased risk of spontaneous abortion, low birth weight, prematurity or congenital malformations has been observed...
March 22, 2017: Expert Review of Clinical Pharmacology
https://www.readbyqxmd.com/read/28324042/interleukin-1-antagonism-decreases-cortisol-levels-in-obese-individuals
#14
Sandrine Andrea Urwyler, Philipp Schuetz, Fahim Ebrahimi, Marc Y Donath, Mirjam Christ-Crain
Context: Increased cortisol levels in obesity may contribute to the associated metabolic syndrome. In obesity, the activated innate immune system leads to increased interleukin (IL)-1β, which is known to stimulate the release of adrenocorticotropin hormone (ACTH). Objective: We hypothesised that in obesity IL-1 antagonism would result in downregulation of the hypothalamo-pituitary-adrenal (HPA) axis, leading to decreased cortisol levels. Design and participants: In this prospective intervention study we included 73 patients with obesity (BMI >30kg/m2) and at least one additional feature of the metabolic syndrome...
February 20, 2017: Journal of Clinical Endocrinology and Metabolism
https://www.readbyqxmd.com/read/28301545/implication-of-the-intestinal-microbiome-as-a-potential-surrogate-marker-of-immune-responsiveness-to-experimental-therapies-in-autoimmune-diabetes
#15
James C Needell, Charles A Dinarello, Diana Ir, Charles E Robertson, Sarah M Ryan, Miranda E Kroehl, Daniel N Frank, Danny Zipris
Type 1 diabetes (T1D) is an autoimmune proinflammatory disease with no effective intervention. A major obstacle in developing new immunotherapies for T1D is the lack of means for monitoring immune responsiveness to experimental therapies. The LEW1.WR1 rat develops autoimmunity following infection with the parvovirus Kilham rat virus (KRV) via mechanisms linked with activation of proinflammatory pathways and alterations in the gut bacterial composition. We used this animal to test the hypothesis that intervention with agents that block innate immunity and diabetes is associated with a shift in the gut microbiota...
2017: PloS One
https://www.readbyqxmd.com/read/28299830/repeated-water-avoidance-stress-induces-visceral-hypersensitivity-role-of-il-1-il-6-and-peripheral-corticotropin-releasing-factor
#16
Tsukasa Nozu, Saori Miyagishi, Rintaro Nozu, Kaoru Takakusaki, Toshikatsu Okumura
BACKGROUND AND AIM: Repeated water avoidance stress (WAS) induces visceral hypersensitivity. Additionally, it is also known to activate corticotropin-releasing factor (CRF), mast cells and pro-inflammatory cytokines systems, but their precise roles on visceral sensation have not been determined definitely. The aim of the study was to explore this issue. METHODS: Abdominal muscle contractions induced by colonic balloon distention, i.e. visceromotor response (VMR) was detected electrophysiologically in conscious rats...
March 15, 2017: Journal of Gastroenterology and Hepatology
https://www.readbyqxmd.com/read/28298024/reduction-of-inflammation-after-administration-of-interleukin-1-receptor-antagonist-following-aneurysmal-subarachnoid-hemorrhage-results-of-the-subcutaneous-interleukin-1ra-in-sah-scil-sah-study
#17
James Galea, Kayode Ogungbenro, Sharon Hulme, Hiren Patel, Sylvia Scarth, Margaret Hoadley, Karen Illingworth, Catherine J McMahon, Nikolaos Tzerakis, Andrew T King, Andy Vail, Stephen J Hopkins, Nancy Rothwell, Pippa Tyrrell
OBJECTIVE Aneurysmal subarachnoid hemorrhage (aSAH) is a devastating cerebrovascular event with long-term morbidity and mortality. Patients who survive the initial bleeding are likely to suffer further early brain injury arising from a plethora of pathological processes. These may result in a worsening of outcome or death in approximately 25% of patients and may contribute to longer-term cognitive dysfunction in survivors. Inflammation, mediated by the cytokine interleukin-1 (IL-1), is an important contributor to cerebral ischemia after diverse forms of brain injury, including aSAH...
February 24, 2017: Journal of Neurosurgery
https://www.readbyqxmd.com/read/28296747/adult-onset-still-s-disease-with-atypical-cutaneous-manifestations
#18
Francisco Javier Narváez Garcia, María Pascual, Mercè López de Recalde, Pablo Juarez, Isabel Morales-Ivorra, Jaime Notario, Anna Jucglà, Joan M Nolla
The diagnosis of adult-onset Still's disease (AOSD) can be very difficult. There are no specific tests available, and diagnosis is usually based on a symptom complex and the well-described typical evanescent rash seen in the majority of patients. However, in recent years, other atypical cutaneous manifestations of AOSD have been reported. These atypical skin eruptions often present in addition to the typical evanescent rash but may also be the only skin manifestation, resulting in delayed diagnosis because of under-recognition...
March 2017: Medicine (Baltimore)
https://www.readbyqxmd.com/read/28293457/anakinra-use-during-pregnancy-report-of-a-case-with-familial-mediterranean-fever-and-infertility
#19
Ufuk İlgen, Orhan Küçükşahin
Familial Mediterranean fever (FMF), affecting people of Mediterranean origin, is an endemic and sometimes problematic disease because of colchicine resistance/intolerance, with relative lack of treatment alternatives, and disease- or treatment-related issues, such as subfertility. Anakinra, being a rational and effective treatment alternative, has no conclusive human pregnancy data. Here we report a case of FMF with infertility who became pregnant with in vitro fertilization (IVF) under treatment with anakinra, along with the pregnancy outcome...
March 2017: European Journal of Rheumatology
https://www.readbyqxmd.com/read/28288653/inflammatory-bowel-disease-following-anti-interleukin-1-treatment-in-systemic-juvenile-idiopathic-arthritis
#20
Boris Hügle, Fabian Speth, Johannes-Peter Haas
BACKGROUND: Inflammatory bowel disease can develop in the context of some rheumatic diseases in childhood, including juvenile idiopathic arthritis (JIA). Inflammatory bowel disease (IBD) is frequently associated with other immune-mediated diseases; however, systemic onset JIA (sJIA) has not previously been connected to IBD. Treatment of sJIA has significantly changed in recent years, possibly causing changes in inflammatory patterns. Therefore, data from the German Center for Pediatric and Adolescent Rheumtology from 2010 until 2015 were analyzed by retrospective chart review...
March 14, 2017: Pediatric Rheumatology Online Journal
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