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macrophage activation syndrome

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https://www.readbyqxmd.com/read/28630236/european-evidence-based-recommendations-for-diagnosis-and-treatment-of-childhood-onset-systemic-lupus-erythematosus-the-share-initiative
#1
Noortje Groot, Nienke de Graeff, Tadej Avcin, Brigitte Bader-Meunier, Paul Brogan, Pavla Dolezalova, Brian Feldman, Isabelle Kone-Paut, Pekka Lahdenne, Stephen D Marks, Liza McCann, Seza Ozen, Clarissa Pilkington, Angelo Ravelli, Annet van Royen-Kerkhof, Yosef Uziel, Bas Vastert, Nico Wulffraat, Sylvia Kamphuis, Michael W Beresford
Childhood-onset systemic lupus erythematosus (cSLE) is a rare, multisystem and potentially life-threatening autoimmune disorder with significant associated morbidity. Evidence-based guidelines are sparse and management is often based on clinical expertise. SHARE (Single Hub and Access point for paediatric Rheumatology in Europe) was launched to optimise and disseminate management regimens for children and young adults with rheumatic diseases like cSLE. Here, we provide evidence-based recommendations for diagnosis and treatment of cSLE...
June 19, 2017: Annals of the Rheumatic Diseases
https://www.readbyqxmd.com/read/28629255/hemophagocytic-lymphohistiocytosis-associated-to-haemophilus-parainfluenzae-endocarditis-a-case-report
#2
D I Costescu Strachinaru, M Chaumont, D Gobin, L Sattar, M Strachinaru, E Karakike, A Roman, D Konopnicki
BACKGROUND: Hemophagocytic lymphohistiocytosis (HLH) is a rare but severe and potentially fatal syndrome that is characterized by increased proliferation and activation of benign macrophages with hemophagocytosis throughout the reticuloendothelial system. This syndrome is classified as primary (genetic) or secondary when acquired in the context of infections (usually viral), malignancies, rheumatologic and metabolic diseases. CASE SUMMARY: We report a case of HLH complicating a Haemophilus parainfluenzae mitral valve endocarditis and resolving under antibiotic and surgical treatment alone...
June 19, 2017: Acta Clinica Belgica
https://www.readbyqxmd.com/read/28614216/refractory-adult-onset-still-disease-complicated-by-macrophage-activation-syndrome-and-acute-myocarditis-a-case-report-treated-with-high-doses-8%C3%A2-mg-kg-d-of-anakinra
#3
Federico Parisi, Annamaria Paglionico, Valentina Varriano, Gianfranco Ferraccioli, Elisa Gremese
RATIONALE: Myocarditis is a rare but potentially fatal complication of Still's disease (about 7% of total cases). PATIENT CONCERNS: A 42-year-old woman was admitted to our ward with high-grade fever, rash and polyarthralgia, lasting since 4 weeks and rapidly complicated by MAS and acute heart failure. DIAGNOSES: Adult Onset Still's Disease rapidly developping macrophage activation syndrome and disseminated intravascular coagulopathy, further complicated by iperacute myocarditis with cardiac arrest...
June 2017: Medicine (Baltimore)
https://www.readbyqxmd.com/read/28613152/reduction-of-infection-by-inhibiting-mtor-pathway-is-associated-with-reversed-repression-of-type-i-interferon-by-porcine-reproductive-and-respiratory-syndrome-virus
#4
Qinfang Liu, Laura C Miller, Frank Blecha, Yongming Sang
Type I interferons (IFNs) are critical in animal antiviral regulation. IFN-mediated signalling regulates hundreds of genes that are directly associated with antiviral, immune and other physiological responses. The signalling pathway mediated by mechanistic target of rapamycin (mTOR), a serine/threonine kinase regulated by IFNs, is key in regulation of cellular metabolism and was recently implicated in host antiviral responses. However, little is known about how animal type I IFN signalling coordinates immunometabolic reactions during antiviral defence...
June 14, 2017: Journal of General Virology
https://www.readbyqxmd.com/read/28608285/the-glp-1-analogue-lixisenatide-decreases-atherosclerosis-in-insulin-resistant-mice-by-modulating-macrophage-phenotype
#5
Ángela Vinué, Jorge Navarro, Andrea Herrero-Cervera, Marta García-Cubas, Irene Andrés-Blasco, Sergio Martínez-Hervás, José T Real, Juan F Ascaso, Herminia González-Navarro
AIMS/HYPOTHESIS: Recent clinical studies indicate that glucagon-like peptide-1 (GLP-1) analogues prevent acute cardiovascular events in type 2 diabetes mellitus but their mechanisms remain unknown. In the present study, the impact of GLP-1 analogues and their potential underlying molecular mechanisms in insulin resistance and atherosclerosis are investigated. METHODS: Atherosclerosis development was evaluated in Apoe (-/-) Irs2 (+/-) mice, a mouse model of insulin resistance, the metabolic syndrome and atherosclerosis, treated with the GLP-1 analogues lixisenatide or liraglutide...
June 12, 2017: Diabetologia
https://www.readbyqxmd.com/read/28607110/the-bile-acid-receptor-gpbar1-regulates-the-m1-m2-phenotype-of-intestinal-macrophages-and-activation-of-gpbar1-rescues-mice-from-murine-colitis
#6
Michele Biagioli, Adriana Carino, Sabrina Cipriani, Daniela Francisci, Silvia Marchianò, Paolo Scarpelli, Daniele Sorcini, Angela Zampella, Stefano Fiorucci
GPBAR1 (TGR5 or M-BAR) is a G protein-coupled receptor for secondary bile acids that is highly expressed in monocytes/macrophages. In this study, we aimed to determine the role of GPBAR1 in mediating leukocyte trafficking in chemically induced models of colitis and investigate the therapeutic potential of BAR501, a small molecule agonist for GPBAR1. These studies demonstrated that GPBAR1 gene ablation enhanced the recruitment of classically activated macrophages in the colonic lamina propria and worsened the severity of inflammation...
June 12, 2017: Journal of Immunology: Official Journal of the American Association of Immunologists
https://www.readbyqxmd.com/read/28598057/macrophage-activation-syndrome
#7
P Babu Raj, B L Harikrishnan, Roshan Mampilly, R Anand
Macrophage Activation Syndrome (MAS) occurs as a severe life-threatening complication of several chronic rheumatic diseases. It is more frequent with systemic onset juvenile arthritis and adult onset Still's disease.1 It can be primary, infection related, malignancy associated or autoimmune3. We report a case of Macrophage Activation Syndrome presenting as pyrexia of unknown origin (PUO) and pancytopenia in the absence of any known triggering factor.
May 2017: Journal of the Association of Physicians of India
https://www.readbyqxmd.com/read/28594137/increased-adipocyte-size-macrophage-infiltration-and-adverse-local-adipokine-profile-in-perirenal-fat-in-cushing-s-syndrome
#8
Sean H P P Roerink, Margreet A E M Wagenmakers, Johan F Langenhuijsen, Dov B Ballak, Hanne M M Rooijackers, Frank C d'Ancona, François M van Dielen, Jan W A Smit, Theo S Plantinga, Romana T Netea-Maier, Ad R M M Hermus
OBJECTIVE: To analyze changes in fat cell size, macrophage infiltration, and local adipose tissue adipokine profiles in different fat depots in patients with active Cushing's syndrome. METHODS: Subcutaneous (SC) and perirenal (PR) adipose tissue of 10 patients with Cushing's syndrome was compared to adipose tissue of 10 gender-, age-, and BMI-matched controls with regard to adipocyte size determined by digital image analysis on hematoxylin and eosin stainings, macrophage infiltration determined by digital image analysis on CD68 stainings, and adipose tissue leptin and adiponectin levels using fluorescent bead immunoassays and ELISA techniques...
June 8, 2017: Obesity
https://www.readbyqxmd.com/read/28592489/sustained-notch2-signaling-in-osteoblasts-but-no-in-osteoclasts-is-linked-to-osteopenia-in-a-mouse-model-of-hajdu-cheney-syndrome
#9
Stefano Zanotti, Jungeun Yu, Archana Sanjay, Lauren Schilling, Chris Schoenherr, Aris N Economides, Ernesto Canalis
Individuals with Hajdu-Cheney syndrome (HCS) present with osteoporosis, and HCS is associated with NOTCH2 mutations causing deletions of the proline-, glutamic acid-, serine-, and threonine-rich (PEST) domain that are predicted to enhance NOTCH2 stability and cause gain-of-function. Previously, we demonstrated that mice harboring Notch2 mutations analogous to those in HCS (Notch2HCS) are severely osteopenic because of enhanced bone resorption. We attributed this phenotype to osteoclastic sensitization to receptor activator of nuclear factor-κB ligand and increased osteoblastic tumor necrosis factor superfamily member 11 (Tnfsf11) expression...
June 7, 2017: Journal of Biological Chemistry
https://www.readbyqxmd.com/read/28589388/-efficacy-and-safety-analysis-of-off-label-treatment-with-biologics-in-autoinflammatory-diseases-experiences-from-a%C3%A2-german-registry%C3%A2-graid2
#10
F Proft, M Fleck, C Fiehn, H Schulze-Koops, M Witt, T Dörner, J C Henes
OBJECTIVE: To evaluate the safety and efficacy of therapy with biologics in patients with autoinflammatory diseases (AIF) or macrophage activating syndrome (MAS) in a real-life setting in Germany. METHODS: The German Register of Autoimmune Diseases 2 (GRAID2) is a retrospective, non-interventional, multicenter registry collecting data from all patients with inflammatory rheumatic diseases refractory to conventional therapy and treated with initial off-label biologics between August 2006 and December 2013...
June 6, 2017: Zeitschrift Für Rheumatologie
https://www.readbyqxmd.com/read/28588173/macrophage-activation-syndrome-in-paediatric-rheumatic-diseases
#11
M I Islam, M K Talukder, M M Islam, K Laila, S A Rahman
Macrophage activation syndrome (MAS) is a potentially fatal complication of rheumatic disorders, which commonly occurs in systemic juvenile idiopathic arthritis (sJIA).This study was carried out with the aims of describing the clinical features, laboratory findings and outcomes of MAS associated with paediatric rheumatic diseases in the Department of Paediatrics, Bangabandhu Sheikh Mujib Medical University (BSMMU) and compare these results with previous studies on MAS. This retrospective study was conducted in the paediatric rheumatology wing of the Department of Paediatrics, Bangabandhu Sheikh Mujib Medical University (BSMMU), Dhaka, Bangladesh...
April 2017: Mymensingh Medical Journal: MMJ
https://www.readbyqxmd.com/read/28587749/pluripotent-stem-cell-models-of-blau-syndrome-reveal-an-ifn-%C3%AE-dependent-inflammatory-response-in-macrophages
#12
Sanami Takada, Naotomo Kambe, Yuri Kawasaki, Akira Niwa, Fumiko Honda-Ozaki, Kazuki Kobayashi, Mitsujiro Osawa, Ayako Nagahashi, Katsunori Semi, Akitsu Hotta, Isao Asaka, Yasuhiro Yamada, Ryuta Nishikomori, Toshio Heike, Hiroyuki Matsue, Tatsutoshi Nakahata, Megumu K Saito
BACKGROUND: Blau syndrome, or early-onset sarcoidosis, is a juvenile-onset systemic granulomatosis associated with a mutation in nucleotide-binding oligomerization domain 2 (NOD2). The underlying mechanisms of Blau syndrome leading to autoinflammation are still unclear, and there is currently no effective specific treatment for Blau syndrome. OBJECTIVES: To elucidate the mechanisms of autoinflammation in patients with Blau syndrome, we sought to clarify the relation between disease-associated mutant NOD2 and the inflammatory response in human samples...
June 3, 2017: Journal of Allergy and Clinical Immunology
https://www.readbyqxmd.com/read/28585210/adiponectin-resistance-in-obesity
#13
Atilla Engin
The decrease in adiponectin levels are negatively correlated with chronic subclinical inflammation markers in obesity. The hypertrophic adipocytes cause obesity-linked insulin resistance and metabolic syndrome. Furthermore, macrophage polarization is a key determinant regulating adiponectin receptor (AdipoR1/R2) expression and differential adiponectin-mediated macrophage inflammatory responses in obese individuals. In addition to decrease in adiponectin concentrations, the decline in AdipoR1/R2 mRNA expression leads to a decrement in adiponectin binding to cell membrane, and this turns into attenuation in the adiponectin effects...
2017: Advances in Experimental Medicine and Biology
https://www.readbyqxmd.com/read/28584972/relation-between-thoracic-aortic-inflammation-and-features-of-plaque-vulnerability-in-the-coronary-tree-in-patients-with-non-st-segment-elevation-acute-coronary-syndrome-undergoing-percutaneous-coronary-intervention-an-fdg-positron-emission-tomography-and-optical
#14
Nevio Taglieri, Cristina Nanni, Gabriele Ghetti, Rachele Bonfiglioli, Francesco Saia, Maria Letizia Bacchi Reggiani, Giacomo Maria Lima, Valeria Marco, Francesco Prati, Stefano Fanti, Claudio Rapezzi
PURPOSE: To evaluate the relationship between aortic inflammation as assessed by (18)F-fluorodeoxyglucose-positron emission tomography ((18)F-FDG-PET) and features of plaque vulnerability as assessed by frequency domain-optical coherence tomography (FD-OCT). METHODS: We enrolled 30 consecutive non-ST-segment elevation acute coronary syndrome patients undergoing percutaneous coronary intervention. All patients underwent three-vessel OCT before intervention and (18)F-FDG-PET before discharge...
June 6, 2017: European Journal of Nuclear Medicine and Molecular Imaging
https://www.readbyqxmd.com/read/28584460/hemophagocytic-lymphohistiocytosis-associated-with-anaplasmosis
#15
Tamara M Johnson, Melinda S Brown, Mohamed Rabbat, Jihad Slim
Hemophagocytic lymphohistiocytosis (HLH) is a hyperinflammatory syndrome characterized by unregulated macrophage and T-lymphocyte activation resulting in cytokine overproduction and subsequent histiocytic phagocytosis. Variant infections, particularly viruses have been postulated as the inciting factor for this potentially fatal disease. Herein, we will report a case of HLH associated with anaplasmosis.
April 2017: Journal of Global Infectious Diseases
https://www.readbyqxmd.com/read/28583781/-macrophage-activation-syndrome-and-kawasaki-disease-four-new-cases
#16
B Dumont, P Jeannoel, L Trapes, E Rolland, C Gay, J-L Stephan
Macrophage activation syndrome (MAS) is a rarely reported complication of Kawasaki disease (KD). It must be sought during KD with unusual clinical signs, such as enlargement of the liver or spleen, cytopenia (including thrombocytopenia), and elevated serum triglycerides. Here, we report four cases from a single center. The first is the description of a case of KD occurring in an 11-month-old child 15 days after the occurrence of infectious spondylitis. For the second, acute myocarditis heralded KD in a 5-year-old child...
June 2, 2017: Archives de Pédiatrie: Organe Officiel de la Sociéte Française de Pédiatrie
https://www.readbyqxmd.com/read/28575810/homology-modeling-and-molecular-docking-studies-on-type-ii-diabetes-complications-reduced-ppar%C3%AE-receptor-with-various-ligand-molecules
#17
S Prabhu, S Vijayakumar, P Manogar, Gaanty Pragas Maniam, Natanamurugaraj Govindan
Peroxisome proliferator-activated receptor gamma (PPARγ), a type II nuclear receptor present in adipose tissue, colon and macrophages. It reduces the hyperglycemia associated metabolic syndromes. Particularly, type II diabetes-related cardiovascular system risk in human beings. The fatty acid storage and glucose metabolism are regulated by PPARγ activation in human body. According to recent reports commercially available PPARγ activating drugs have been causing severe side effects. At the same time, natural products have been proved to be a promising area of drug discovery...
May 30, 2017: Biomedicine & Pharmacotherapy, Biomédecine & Pharmacothérapie
https://www.readbyqxmd.com/read/28566469/visceral-adipose-tissue-activated-macrophage-content-and-inflammatory-adipokine-secretion-is-higher-in-pre-eclampsia-than-in-healthy-pregnancy
#18
Shahzya Huda, FIona Jordan, JAck Bray, Gillian Love, Reba Payne, Naveed Sattar, Dilys J Freeman
Obesity increases preeclampsia risk. Adipose tissue inflammation may contribute to the clinical syndrome of pre-eclampsia. We compared adipose tissue macrophage infiltration and release of pro-inflammatory adipokines in pre-eclampsia and healthy pregnancy. Subcutaneous and visceral adipose tissue biopsies were collected from healthy (n=13) and preeclampsia (n=13) mothers. Basal and lipopolysaccharide stimulated adipocyte TNFα, IL-6, CCL-2 and CRP release was measured. Adipose tissue cell densities of activated (cfms(+)) and total (CD68(+)) macrophages were determined...
May 31, 2017: Clinical Science (1979-)
https://www.readbyqxmd.com/read/28562511/kawasaki-disease-complicated-with-macrophage-activation-syndrome-a-systematic-review
#19
Susana García-Pavón, Marco A Yamazaki-Nakashimada, Milton Báez, Karla L Borjas-Aguilar, Chiharu Murata
Macrophage activation syndrome (MAS), also known as secondary hemophagocytic lymphohistiocytosis, is a rare and potentially fatal complication of Kawasaki disease (KD). We report 2 cases, performed a literature search, and analyze the characteristics of MAS associated with KD. A total of 69 patients were evaluated, 34 reported the date of the diagnosis of MAS and KD, 6% had a diagnosis of MAS before KD, 21% had a simultaneous presentation, and 73% had the diagnosis of MAS after KD. Different treatment approaches were observed with corticosteroids administered in 87%, cyclosporine in 49%, etoposide (VP-16) in 39%, and monoclonal anti-TNF in 6% of cases...
May 29, 2017: Journal of Pediatric Hematology/oncology
https://www.readbyqxmd.com/read/28559417/the-ftd-like-syndrome-causing-trem2-t66m-mutation-impairs-microglia-function-brain-perfusion-and-glucose-metabolism
#20
Gernot Kleinberger, Matthias Brendel, Eva Mracsko, Benedikt Wefers, Linda Groeneweg, Xianyuan Xiang, Carola Focke, Maximilian Deußing, Marc Suárez-Calvet, Fargol Mazaheri, Samira Parhizkar, Nadine Pettkus, Wolfgang Wurst, Regina Feederle, Peter Bartenstein, Thomas Mueggler, Thomas Arzberger, Irene Knuesel, Axel Rominger, Christian Haass
Genetic variants in the triggering receptor expressed on myeloid cells 2 (TREM2) increase the risk for several neurodegenerative diseases including Alzheimer's disease and frontotemporal dementia (FTD). Homozygous TREM2 missense mutations, such as p.T66M, lead to the FTD-like syndrome, but how they cause pathology is unknown. Using CRISPR/Cas9 genome editing, we generated a knock-in mouse model for the disease-associated Trem2 p.T66M mutation. Consistent with a loss-of-function mutation, we observe an intracellular accumulation of immature mutant Trem2 and reduced generation of soluble Trem2 similar to patients with the homozygous p...
May 30, 2017: EMBO Journal
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