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Abstracts tagged "macrophage activation syndrome"

  • Abstract Number: 0562 • ACR Convergence 2022

    Large-Scale Targeted Sequencing Study Links Systemic Juvenile Idiopathic Arthritis with Rare Variants of MEFV, LYST, STXBP2, UNC13D

    Mariana Correia Marques1, Danielle Rubin2, Emily Shuldiner2, Elizabeth Schmitz2, Elizabeth Baskin2, Andrew Patt3, Alexei Grom4, Dirk Foell5, Marco Gattorno6, John Bohnsack7, Rae Yeung8, Sampath Prahalad9, Elizabeth Mellins10, Jordi Antón11, Claudio Len12, Sheila Oliveira13, Patricia Woo14, Seza Ozen15, INCHARGE Consortium16 and Michael Ombrello17, 1National Institute of Arthritis and Musculoskeletal and Skin Diseases / Children`s National Hospital, Bethesda, MD, 2National Institute of Arthritis and Musculoskeletal and Skin Diseases, Bethesda, MD, 3National Center for Advancing Translational Sciences, Bethesda, MD, 4Divisions of Rheumatology, Cincinnati Children’s Hospital Medical Center, Cincinnati, OH, 5University Hospital Münster, Münster, Germany, 6Pediatric Clinic and Rheumatology Unit, IRCCS Istituto Giannina Gaslini, Genoa, Italy, 7University of Utah, Salt Lake City, UT, 8The Hospital for Sick Children, University of Toronto, Toronto, ON, Canada, 9Emory + Children's Pediatric Institute, Atlanta, GA, 10Stanford University, Stanford, CA, 11Pediatric Rheumatology Department. Hospital Sant Joan de Déu. Universitat de Barcelona, Esplugues de Llobregat, Spain, 12Universidade Federal de São Paulo, São Paulo, Brazil, 13Universidade Federal do Rio de Janeiro, Rio De Janeiro, Brazil, 14University College London, London, United Kingdom, 15Hacettepe University Faculty of Medicine, Ankara, Turkey, 16International Childhood Arthritis Genetics Consortium, Bethesda, MD, 17National Institutes of Arthritis and Musculoskeletal and Skin Diseases (NIAMS), National Institutes of Health (NIH), Bethesda, MD

    Background/Purpose: Systemic juvenile idiopathic arthritis (sJIA) is a genetically complex inflammatory condition. It can be marked by severe systemic inflammation that resembles the hereditary periodic…
  • Abstract Number: 0569 • ACR Convergence 2022

    Cytokine Storm Susceptibility Factors IL-18 and Cytotoxic Impairment Converge on Pathologic CD8 T-cell Hyperactivation

    Emily Landy1, Scott Canna2, Paul Tsoukas3, Vinh Dang2 and Jemy Varghese2, 1University of Pittsburgh, Pittsburgh, PA, 2Children's Hospital of Philadelphia, Philadelphia, PA, 3The Hospital for Sick Children, Toronto, ON, Canada

    Background/Purpose: Mounting evidence suggests partial defects in cytotoxicity-related genes may promote hyperinflammation in Macrophage Activation Syndrome (MAS) patients, who uniformly have highly-elevated IL-18. In mice,…
  • Abstract Number: 0799 • ACR Convergence 2022

    Serum Laboratory Parameters of Hemophagocytic Lymphohistiocytosis Syndrome in Patients with Autoimmune Rheumatic Diseases Infected with SARS-CoV-2: A Retrospective Comparative Study

    Omar Alsaed, Samar Al emadi, Karima Becetti, Eman Satti, Hadil Ashour, Yousef Alrimawi, Miral H.Gharib, Mohamad Alkahlout, Rawan Saleh, Basem Awadh and Mohammed Hammoudeh, Hamad Medical Corporation, Doha, Qatar

    Background/Purpose: The serum features of the cytokine storm induced by severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection are similar to those of hemophagocytic lymphohistiocytosis…
  • Abstract Number: 0825 • ACR Convergence 2022

    Predicting Outcomes in Macrophage Activation Syndrome (MAS): A Retrospective Study on 43 Consecutive Patients with MAS

    Sumanth Madan1, Suma Balan1, MITHUN CHALAKKARAYIL BHAGAVALDAS2, Spoorthy DR1, Sudeep Rath1, Joslyn Thattil1, Amisha Shahul1 and Niveditha Kartha1, 1Amrita Institute of Medical Sciences, Kochi, India, 2Amrita Institute of Medical Sciences, Muvattupuzha, India

    Background/Purpose: Macrophage activation syndrome is a diagnosis along the spectrum of hemophagocytic lymphohistiocytosis (HLH) spectrum disorders. It is a rare, yet potentially fatal disease characterized…
  • Abstract Number: 0859 • ACR Convergence 2022

    Systemic Juvenile Idiopathic Arthritis Associated Lung Disease in Europe

    Claudia Bracaglia1, Francesca Minoia2, Christoph Kessel3, Sebastiaan Vastert4, Manuela Pardeo1, Alessia Arduini1, Ozge Basaran5, Nural kiper6, Mikhail Kostik7, Mia Glerup8, Sarka Fingerhutova9, Roberta Caorsi10, AnnaCarin Horne11, Giovanni Filocamo12, Helmut Wittkowski3, Marija Jelusic13, Jordi Anton14, Samira Khaldi-Plassart15, Alexandre Belot16, Gerd Horneff17, Seraina Palmer Sarott18, Elvira cannizzaro Schneider18, Pavla Dolezalova9, Angelo Ravelli19, Seza Ozen20 and Fabrizio De Benedetti1, 1Division of Rheumatology, IRCCS Ospedale Pediatrico Bambino Gesu', Rome, Italy, 2Fondazione IRCCS Ca' Granda Ospedale Maggiore Policlinico, Milano, Italy, 3Department of Pediatric Rheumatology & Immunology, WWU Medical Center (UKM), Münster, Germany, 4Pediatric Rheumatology & Immunology, University Medical Center Utrecht, Utrecht, Netherlands, 5Department of Pediatrics, Division of Pediatric Rheumatology, Hacettepe University, Ankara, Turkey, 6Department of Pediatrics, Division of Pediatric Pulmonology, Hacettepe University, Ankara, Turkey, 7Saint-Petersburg State Pediatric Medical University, Saint Petersburg, Russia, 8Department of Pediatrics, Aarhus University Hospital, Aarhus, Denmark, 9Centre for Paediatric Rheumatology and Autoinflammatory Diseases , Department of Paediatrics and Inherited Metabolic Disorders, 1st Faculty of Medicine, Charles University and General University Hospital in Prague, Prague, Czech Republic, 10Department of pediatrics and Rheumatology, IRRCS Istituto G. Gaslini, Genova, Italy, 11Department of pediatric rheumathology Karolinska University Hospital and Department of pediatrics, Karolinska Institute, Stockholm, Sweden, 12UOC Pediatria a Media Intensità di Cure, Clinica de Marchi, Milano, Italy, 13Department of Pediatrics, University of Zagreb School of Medicine, University Hospital Centre Zagreb, Zagreb, Croatia, 14Pediatric Rheumatology, Hospital Sant Joan de Déu, Universitat de Barcelona, Barcelona, Spain, 15Pediatric Nephrology, Rheumatology, Dermatology Unit, Hôpital Femme Mère Enfant, Hospices Civils de Lyon, Lyon, France, 16Hospices Civils de Lyon, Collonges au mont d'or, France, 17Pediatrics, Asklepios Klinik Sankt Augustin GmbH, Sankt Augustin, Germany, 18Paediatric Rheumatology University Children’s Hospital Zurich, Zürich, Switzerland, 19Department of Neurosciences, Rehabilitation, Ophthalmology, Genetic and Maternal Infantile Sciences (DINOGMI) University of Genoa, Italy,Scientific Direction, IRCCS Istituto Giannina Gaslini, Genova, Italy, 20Hacettepe University Faculty of Medicine, Ankara, Turkey

    Background/Purpose: Chronic parenchymal lung disease (LD) is a new emerging severe life-threatening complication of sJIA. The number of sJIA patients with LD is apparently increasing…
  • Abstract Number: 0864 • ACR Convergence 2022

    Baseline Clinical and Laboratory Features of the Childhood Arthritis and Rheumatology Research Alliance (CARRA) Systemic Juvenile Idiopathic Arthritis-Associated Lung Disease (SJIA-LD) Cohort

    Esraa Eloseily1, Min-Lee Chang2, MaryEllen Riordan3, Alan Russell4, Marc Natter2, Yukiko Kimura5 and Grant Schulert6, 1Division of Pediatric Rheumatology, Cincinnati Children's Hospital Medical Center, Cincinnati, OH, 2Boston Children's Hospital, Boston, MA, 3Hackensack Meridian Health, Hackensack, NJ, 4Duke Clinical Research Institute, Duke, NC, 5Hackensack Meridian Health, New York, NY, 6Cincinnati Children's Hospital Medical Center, Cincinnati, OH

    Background/Purpose: Systemic juvenile idiopathic arthritis (SJIA) associated lung disease (SJIA-LD) is an emerging and life threatening clinical problem, and currently affects as many as 1…
  • Abstract Number: 0872 • ACR Convergence 2022

    Incidence, Risk Factors, and Outcomes of Eosinophilia on IL-1 and IL-6 Inhibitors in Systemic and Non-Systemic Juvenile Idiopathic Arthritis

    Holly Wobma1, Maria Taylor2, Ki Pui Lam1, Helene Brown1, Siobhan Case1, Mia Chandler1, Margaret Chang1, Ezra Cohen1, Megan Day-Lewis1, Olha Halyabar1, Jonathan Hausmann1, Melissa Hazen3, Erin Janssen1, Pui Lee1, Mindy Lo1, Esra Meidan1, Jordan Roberts1, Mary Beth F. Son1, Robert Sundel1, Fatma Dedeoglu1, Peter Nigrovic1, Alicia Casey4, Joyce Chang1 and lauren henderson1, 1Division of Immunology, Boston Children's Hospital, Boston, MA, 2Division of Immunology, Boston Children's Hospital, Brighton, MA, 3Boston Children's Hospital, Boston, MA, 4Division of Pulmonary Medicine, Boston Children's Hospital, Boston, MA

    Background/Purpose: Children with systemic juvenile idiopathic arthritis (sJIA) exposed to IL1/6 inhibitors may develop eosinophilia with an atypical rash, reportedly in association with HLA-DRB1*15:XX. There…
  • Abstract Number: 2213 • ACR Convergence 2022

    Multi-omic Analysis of Macrophage Activation Syndrome Associated with sJIA Reveals a Potential Role of Type I Interferons in the Expansion of Cycling T Cells

    Kailey Brodeur1, Liang Chen1, zhengping huang2, Yan Du1, Holly Wobma3, Maria Taylor4, Joyce Chang3, Megan Day-Lewis3, Fatma Dedeoglu3, Olha Halyabar3, Mindy Lo3, Jane W. Newburger5, Mary Beth F. Son3, Robert Sundel3, Peter Nigrovic3, lauren henderson3 and Pui Lee3, 1Boston Children's Hospital, Boston, MA, 2Guangdong Second Provincial Hospital, Guangzhou, China, 3Division of Immunology, Boston Children's Hospital, Boston, MA, 4Division of Immunology, Boston Children's Hospital, Brighton, MA, 5Department of Cardiology, Boston Children's Hospital, Harvard Medical School, Boston, MA

    Background/Purpose: Macrophage activation syndrome (MAS) is a complication of systemic juvenile idiopathic arthritis (sJIA) characterized by cytokine storm and overt immune cell activation. We aim…
  • Abstract Number: L20 • ACR Convergence 2021

    Efficacy of Emapalumab, an Anti-IFNγ Antibody in Patients with Macrophage Activation Syndrome (MAS) Complicating Systemic Juvenile Idiopathic Arthritis (sJIA) Who Had Failed High-Dose Glucocorticoids (GCs)

    Fabrizio De Benedetti1, Alexei Grom2, Paul Brogan3, Claudia Bracaglia1, Manuela Pardeo1, Giulia Marucci1, Despina Eleftheriou3, Charalampia Papadopoulou3, Pierre Quartier4, Jordi Antón5, Rikke Frederiksen6, Veronica Asnaghi6 and Cristina De Min6, 1Bambino Gesù Children's Hospital IRCCS, Rome, Italy, 2Cincinnati Children’s Hospital, Division of Rheumatology, Cincinnati, OH, 3UCL Institute of Child Health, and Great Ormond Street Hospital NHS Foundation Trust, London, United Kingdom, 4Université de Paris, IMAGINE Institute, RAISE reference centre, Pediatric Immuno-Hematology and Rheumatology Unit, Necker Hospital, Assistance Publique-Hôpitaux de Paris, Paris, France, 5Hospital Sant Joan de Déu, Division of Pediatric Rheumatology, University of Barcelona, Barcelona, Spain, 6Swedish Orphan Biovitrum AG (Sobi), Basel, Switzerland

    Background/Purpose: MAS is a life-threatening complication of rheumatic diseases, occurring most frequently in sJIA. The mainstay of MAS treatment is high dose GCs; however, GCs…
  • Abstract Number: 1928 • ACR Convergence 2021

    In Vitro and In Vivo Evidence for DOCK8 as a Risk Allele for Cytokine Storm Syndrome, Including COVID-19 and MIS-C

    Randy Cron1, Mingce Zhang1, Niansheng Chu2, Devin Absher3, John Bridges1, Amanda Schnell1, Anshul Vagrecha4, Shannon Lozinsky4, Suchitra Acharya4, Carolyn Levy4, Winn Chatham1 and Edward Behrens2, 1University of Alabama at Birmingham, Birmingham, AL, 2University of Pennsylvania, Philadelphia, PA, 3HudsonAlpha Institute for Biotechnology, Huntsville, AL, 4Hofstra/Northwell School of Medicine, Hempstead, NY

    Background/Purpose: Cytokine storm syndromes (CSS) are frequently fatal complications of a variety of oncologic, rheumatic, and infectious diseases. Many patients with CSS possess heterozygous missense…
  • Abstract Number: 1930 • ACR Convergence 2021

    Single-Cell Genomics Reveals a Shared Monocyte Interferon Program in a Subset of Patients with Systemic Juvenile Idiopathic Arthritis, Macrophage Activation Syndrome and Lung Disease

    Emely Verweyen1, Kairavee Thakkar2, Kashish Chetal2, Sanjeev Dhakal3, Alexei Grom2, Nathan Salomonis2 and Grant Schulert2, 1Cincinnati Children's Hospital, Cincinnati, OH, 2Cincinnati Children's Hospital Medical Center, Cincinnati, OH, 3Cincinnati Children hospital, Cincinnati, OH

    Background/Purpose: Systemic juvenile idiopathic arthritis (SJIA) is a clinically heterogenous disease and can be complicated by macrophage activation syndrome (MAS) and lung disease (LD) thought…
  • Abstract Number: 0196 • ACR Convergence 2021

    A Single Center, Double Blind, Randomized, Placebo-Controlled Trial of Anakinra in Adult Patients with Features of Cytokine Storm Syndrome in COVID-19

    Lesley Jackson1, Randy Cron1, Nitasha Khullar2, Christopher Chapleau3, Dongmei Sun4 and Winn Chatham1, 1University of Alabama at Birmingham, Birmingham, AL, 2University of Alabama Birmingham, Birmingham, AL, 3UAB Pharmacy, University of Alabama at Birmingham, Birmingham, AL, 4Division of Clinical Immunology and Rheumatology, The University of Alabama at Birmingham, Birmingham, AL

    Background/Purpose: Some patients with COVID-19 develop respiratory distress and cytokine storm syndrome (CSS) which is characterized by hyperinflammation and may progress to multi-organ failure. Anakinra…
  • Abstract Number: 0246 • ACR Convergence 2021

    Macrophage Activating Syndrome in Patients with Systemic Juvenile Idiopathic Arthritis Treated with Biological Drugs

    Betul Sozeri1, Kenan Barut2, Erdal Atalay3, Aysenur Pac Kisaarslan4, Semanur Ozdel5, Ozge Altug6, Ferhat Demir1, Balahan Makay7, Nuray Aktay Ayaz8, Ersen Acar9, Fatih Haslak2, Erdal Sag10, Mehmet YILDIZ2, Ummusen Kaya Akca10, Amra Adrovic2, Yelda Bilginer10, Hakan Poyrazoglu11, Erbil Unsal7, Ozgur Kasapcopur12 and Seza Ozen3, 1University of Health Sciences, Umraniye Tranining and Research Hospital Division of Pediatric Rheumatology, Istanbul, Turkey, 2Istanbul University-Cerrahpasa, Faculty of Medicine, Pediatric Rheumatology, İstanbul, Turkey, 3Hacettepe University Faculty of Medicine, Pediatric Romatology, Ankara, Turkey, 4Erciyes University Medical Faculty, Pediatric Rheumatology, Kayeri, Turkey, 5Ankara Dr.Sami Ulus Gynecology and Pediatrics Research and Training Hospital, Pediatric Rheumatology, Ankara, Turkey, 6Dr Behcet Uz Children's Hospital, Pediatric Rheumatology, Izmir, Turkey, 7Dokuz Eylul University Faculty of Medicine, Pediatric Rheumatology, Izmir, Turkey, 8Istanbul University, Faculty of Medicine, Pediatric Rheumatology, İstanbul, Turkey, 9Novartis, Istanbul, Turkey, 10Hacettepe University, Division of Pediatric Rheumatology, Department of Pediatrics, Faculty of Medicine, Ankara, Turkey, 11Erciyes University Faculty of Medicine, Pediatric Rheumatology, Kayseri, Turkey, 12Istanbul University-Cerrahpasa, Cerrahpasa Medical School, İstanbul, Turkey

    Background/Purpose: Systemic Juvenile Idiopathic Arthritis (SJIA) is the most acute and severe form of JIA. Despite the substantial evidence supporting the effectiveness of biologic drugs…
  • Abstract Number: 0252 • ACR Convergence 2021

    Role of Clinical and Laboratory Parameters in Differentiating Infection from Disease Flare in Febrile Patients of Systemic Juvenile Idiopathic Arthritis

    Rajesh Kanumuri1, Suma Balan2, Vishal Marwaha2, Sajitha Krishnan2 and Pranav Chickermane2, 1Amrita Institute of Medical Sciences, Guntur, India, 2Amrita Institute of Medical Sciences, Kochi, India

    Background/Purpose: Fever is the most common presentation of systemic juvenile idiopathic arthritis (SJIA) and it is difficult to predict whether the fever is due to…
  • Abstract Number: 0254 • ACR Convergence 2021

    Patient and Disease-Level Factors Associated with Sustained Cessation of Medication for Disease Remission in Systemic Juvenile Idiopathic Arthritis

    Elaine Flanagan1, Rosemary Peterson2, Susan Shenoi3, Helena Chang4, Kelly Wang4, Rebecca Trachtman4 and Karen Onel5, 1Emory/CHOA, Atlanta, GA, 2Dell Children's Medical Center, Austin, TX, 3Seattle Children's Hospital, Seattle, WA, 4Icahn School of Medicine at Mount Sinai, New York, NY, 5Hospital for Special Surgery, New York, NY

    Background/Purpose: The emergence of IL-1 and IL-6 inhibitors (biologics) for the treatment of systemic juvenile idiopathic arthritis (SJIA) has dramatically improved patient outcomes. With higher…
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