Abstracts tagged "macrophage activation syndrome"

Abstract Number: 1711 • ACR Convergence 2023
CD14+ Monocytes Demonstrate a Unique Transcriptional Signature in Macrophage Activation Syndrome, Highlighting a Role for Interferons and Identifying Putative Hemophagocytes in Circulation
Susan Canny¹, Hannah DeBerg², Griffin Gessay², Ailing Lu³, Mary Eckert⁴, Andrea La Bella⁵, Susan Shenoi⁶, Joyce Hui-Yuen⁷, Betsy Barnes⁸ and Jessica Hamerman², ¹Seattle Children's Hospital/University of Washington, Seattle, WA, ²Benaroya Research Institute, Seattle, WA, ³Feinstein Institutes for Medical Research, Manhasset, NY, ⁴Seattle Children's Hospital, Seattle, WA, ⁵Cohen Children's Medical Center, Queens, NY, ⁶Seattle Childrens Hospital, Mercer Island, WA, ⁷Division of Pediatric Rheumatology, Cohen Children's Medical Center, Lake Success, NY, ⁸Northwell Health, Manhasset, NY
Background/Purpose: Macrophage activation syndrome (MAS) is a potentially fatal complication of rheumatic diseases. MAS is characterized by a dysfunctional hyperinflammatory response in which there is…
Abstract Number: 2473 • ACR Convergence 2023
A Consensus Based Algorithm to Screen for Lung Disease in Children with Systemic Juvenile Idiopathic Arthritis
Holly Wobma¹, Ronny Bachrach², Joseph Farrell², Margaret Chang³, Megan Day-Lewis¹, Fatma Dedeoglu¹, Martha Fishman⁴, Olha Halyabar¹, Claudia Harris¹, Daniel Ibanez¹, Liyoung Kim¹, Timothy Klouda⁴, Katie Krone⁴, Pui Lee¹, Mindy Lo¹, Kyle McBrearty¹, Esra Meidan¹, Susan Prockop⁵, Aaida Samad⁴, Mary Beth Son¹, Peter Nigrovic³, Alicia Casey⁴, Joyce Chang³ and Lauren Henderson³, ¹Division of Immunology, Boston Children's Hospital, Boston, MA, ²Community Member, Boston, MA, ³Boston Children's Hospital, Boston, MA, ⁴Division of Pulmonary Medicine, Boston Children’s Hospital, Boston, MA, ⁵Division of Hematology-Oncology, Boston Children's Hospital, Boston, MA
Background/Purpose: Over the last decade, lung disease (LD) has become an increasingly recognized complication of systemic juvenile idiopathic arthritis (sJIA). Children with sJIA-LD may be…
Abstract Number: 0039 • ACR Convergence 2023
Enrichment of Rare Variants of Hemophagocytic Lymphohistiocytosis Genes in Systemic Juvenile Idiopathic Arthritis
Mariana Correia Marques¹, Danielle Rubin¹, Emily Shuldiner², Mallika Datta¹, Elizabeth Schmitz¹, Alexei Grom³, Dirk Foell⁴, Marco Gattorno⁵, John Bohnsack⁶, Rae Yeung⁷, Sampath Prahalad⁸, Elizabeth Mellins⁹, Jordi Anton Lopez¹⁰, Claudio Len¹¹, Sheila Oliveira¹², Patricia Woo¹³, Seza Ozen¹⁴, INCHARGE Consortium¹, Zuoming Deng¹⁵ and Michael Ombrello¹, ¹National Institute of Arthritis & Musculoskeletal and Skin Diseases (NIAMS), National Institutes of Health (NIH), Bethesda, MD, ²Department of Biology, Stanford University, Stanford, CA, ³Division of Rheumatology, Cincinnati Children`s Hospital, Cincinnati, OH, ⁴University Hospital Münster, Münster, Germany, ⁵UOC Reumatologia e Malattie Autoinfiammatorie, Genoa, Italy, ⁶University of Utah, Salt Lake City, UT, ⁷The Hospital for Sick Children, University of Toronto, Toronto, ON, Canada, ⁸Emory University School of Medicine, Children's Pediatric Institute, Atlanta, GA, ⁹Stanford University, Stanford, CA, ¹⁰Pediatric Rheumatology Department, Hospital Sant Joan e Deu, Barcelona University, Barcelona, Spain, ¹¹São Paulo Federal University, São Paulo, Brazil, ¹²Universidade Federal do Rio de Janeiro, Rio De Janeiro, Brazil, ¹³University College London, London, United Kingdom, ¹⁴Hacettepe University Medical Faculty, Ankara, Turkey, ¹⁵National Institute of Arthritis and Musculoskeletal and Skin Diseases (NIAMS), National Institutes of Health (NIH), Bethesda, MD
Background/Purpose: Systemic juvenile idiopathic arthritis (sJIA) is a complex inflammatory condition of childhood. It can be complicated by macrophage activation syndrome (MAS), a secondary form…
Abstract Number: 0348 • ACR Convergence 2023
The Use of Ruxolitinib for Improved Disease Control in Systemic Juvenile Idiopathic Arthritis (sJIA) and Recurrent Macrophage Activation Syndrome (MAS)
Kathleen Collins¹, Iman Abutineh¹, Trisha Paul², Parul Rai³, Grant Schulert⁴ and Melissa Hines², ¹University of Tennessee Health Science Center, Memphis, TN, ²St Jude Children's Research Hospital, Memphis, TN, ³St Jude Children's Reseaerch Hospital, Memphis, TN, ⁴Cincinnati Children's Hospital Medical Center, Cincinnati, OH
Background/Purpose: The advent of biologics such as IL-1 receptor antagonists has dramatically improved outcomes for children with pediatric rheumatic diseases and hyperinflammation. However, there remains…
Abstract Number: 0363 • ACR Convergence 2023
Allogenic Hematopoietic Stem Cell Transplant for Children with Refractory Systemic Juvenile Idiopathic Arthritis and sJIA-Associated Lung Disease: 6-month Post-Transplant Outcomes from an International Cohort
Michael Matt¹, Daniel Drozdov¹, Rolla Abu-Arja², Shanmuganathan Chandrakasan³, Kyla Driest², Elvira Cannizzaro Schneider⁴, Despina Moshous⁵, Benedicte Neven⁵, Karen Onel⁶, Sampath Prahalad⁷, Susan Prockop⁸, Pierre Quartier⁵, Johannes Roth⁹, Donna Wall¹⁰, Ulrike Zeilhofer¹¹, Scott Canna¹², Alexei Grom¹, Grant Schulert¹ and Rebecca Marsh¹, ¹Cincinnati Children's Hospital Medical Center, Cincinnati, OH, ²Nationwide Children's Hospital, Columbus, OH, ³Emory University School of Medicine, Atlanta, GA, ⁴Universitaetskinderspital Zurich, Zurich, Switzerland, ⁵Paris Cité Université and Necker Hospital, Paris, France, ⁶Hospital for Special Surgery, New York, NY, ⁷Emory University School of Medicine, Children's Pediatric Institute, Atlanta, GA, ⁸Division of Hematology-Oncology, Boston Children's Hospital, Boston, MA, ⁹University of Ottawa, Ottawa, ON, Canada, ¹⁰The Hospital for Sick Children, Toronto, ON, Canada, ¹¹Children's Hospital of Zurich, Zurich, Switzerland, ¹²Children's Hospital of Philadelphia, Philadelphia, PA
Background/Purpose: Refractory systemic juvenile idiopathic arthritis (sJIA) in children can be complicated by repeated episodes of macrophage activation syndrome (MAS) or sJIA-related lung disease (sJIA-LD).…
Abstract Number: 0378 • ACR Convergence 2023
Characteristics of Macrophage Activation Syndrome in Systemic JIA Patients Receiving Anakinra as First-line Treatment
Remco Erkens¹, Laura Huber¹, Anouk Verwoerd¹, Greta Rogani¹, Dieneke Schonenberg-Meinema², Merlijn van den Berg², Wineke Armbrust³, Elizabeth Legger⁴, Sylvia Kamphuis⁵, Ellen Schatorjé⁶, Esther Hoppenreijs⁷, Joost Swart⁸, Marc Jansen¹, Jorg van Loosdregt¹ and Sebastiaan Vastert¹, ¹University Medical Center Utrecht, Utrecht, Netherlands, ²Emma Children's Hospital, Amsterdam University Medical Centers, Amsterdam, Netherlands, ³University Medical Hospital Groningen, Groningen, Netherlands, ⁴University Medical Center Groningen, Groningen, Netherlands, ⁵Erasmus University Medical Center, Department of Pediatric Rheumatology, Rotterdam, Netherlands, ⁶University Medical Center Radboud, Nijmegen, Netherlands, ⁷Radboud University Medical Center, Nijmegen, Netherlands, ⁸Wilhelmina Children's Hospital / UMC Utrecht, Utrecht, Netherlands
Background/Purpose: Systemic Juvenile Idiopathic Arthritis (sJIA) is a severe inflammatory disease with auto-inflammatory characteristics. The introduction of targeted biologic therapies has revolutionized the treatment and…
Abstract Number: 0758 • ACR Convergence 2023
Clinical and Biological Characteristics of Children and Adults Affected with Still’s Disease: A Systematic Review and Meta-analysis Informing the 2023 EULAR/PReS Recommendations for the Diagnosis and Management of Systemic Juvenile Idiopathic Arthritis and Adult-onset Still’s Disease
STEPHANE MITROVIC¹, Arianna De Matteis², Sara Bindoli³, Fabrizio De Benedetti⁴, Bruno Fautrel⁵, Loreto Carmona⁶ and On Behalf Of The Eular/PreS QoC011 Task Force Member⁷, ¹Pitie Salpetriere Hospital, Sorbonne University, APHP, Paris, France, ²IRCCS Ospedale Pediatrico Bambino Gesu', Rome, Italy, ³Rheumatology Unit, Dept of Medicine, University of Padova, Padova, Italy, ⁴Bambino Gesu Children's Hospital, Division of Rheumatology, Rome, Italy, ⁵Sorbonne Université APHP, Paris, France, ⁶Instituto de Salud Musculoesquelética (InMusc), Madrid, Spain, ⁷EULAR/PReS QoC011 Task Force members are: Jordi Anton, Alexandre Belot, Claudia Bracaglia, Tamas Constantin, Lorenzo Dagna, Alessandro De Bartolo, Eugen Feist, Dirk Foell, Marco Gattorno, Sophie Georgin-Lavialle, Roberto Giacomelli, Alexei Grom, Yvan Jamilloux, Katarina Laskari, Calin Lazar, Francesca Minoia, Peter Nigrovic, Filipa Oliveira Ramos, Seza Ozen, Pierre Quartier-dit-Maire, Piero Ruscitti, Erdal Sag, Sinisa Savic, Marie-Elise Truchetet, Bas Vastert, Tanita Wilhelmer, Carine Wouters., Paris, France
Background/Purpose: Systemic juvenile idiopathic arthritis (sJIA) and adult-onset Still’s disease (AOSD) are part of the non-familial (sporadic) systemic inflammatory disorders and are frequently mentioned as…
Abstract Number: 0759 • ACR Convergence 2023
Derivation and Validation of Four Patient Clusters in Still’s Disease, Results from GIRRCS AOSD-study Group and AIDA Network Still Disease Registry
Piero Ruscitti¹, Antonio Vitale², Ilenia Di Cola¹, Roberto giacomelli³ and Luca Cantarini⁴, ¹University of L'Aquila, L'Aquila, Italy, ²University of Siena, Siena, Italy, ³University of Rome Campus Biomedico, Rome, Italy, ⁴Department of Medical Sciences, Surgery and Neurosciences, Rheumatology Unit, University of Siena and Azienda Ospedaliero-Universitaria Senese [European Reference Network (ERN) for Rare Immunodeficiency, Autoinflammatory and Autoimmune Diseases (RITA) Center], Siena, Italy
Background/Purpose: Still's disease is a rare inflammatory disorder, these patients have a highly heterogeneous disease according to age of onset, clinical presentation, presence of life-threatening…
Abstract Number: 031 • 2023 Pediatric Rheumatology Symposium
Multisystem Inflammatory Syndrome in Children Phenotypes Vary Between SARS-CoV-2 Variants
Greta Mastrangelo¹, Ellen Go², Paul Tsoukas², Hua Lu³, Amy Xu², Arthur Hoi Hin Cheng² and Rae Yeung⁴, ¹The Hospital of Sick Children,Department of Paediatrics, University of Toronto, Division of Rheumatology, Toronto, ON, Canada, ²The Hospital of Sick Children, Department of Paediatrics, University of Toronto, Division of Rheumatology; Cell Biology Program, The Hospital for Sick Children Research Institute, Toronto, ON, Canada, ³The Hospital for Sick Children, Toronto, ON, Canada, ⁴The Hospital for Sick Children, University of Toronto, Toronto, ON, Canada
Background/Purpose: Multisystem Inflammatory Syndrome in Children (MIS-C) is a serious complication associated with COVID-19, presenting as a hyperinflammatory disorder characterized by fever and multiorgan dysfunction.…
Abstract Number: 037 • 2023 Pediatric Rheumatology Symposium
Transcriptional Analysis of CD14+ Monocytes During Macrophage Activation Syndrome Highlights Role for Interferons and RNA Sensing in Monocytes
Susan Canny¹, Hannah DeBerg², Griffin Gessay², Ailing Lu³, Mary Eckert¹, Andrea La Bella⁴, Susan Shenoi⁵, Joyce Hui-Yuen⁶, Betsy Barnes⁷ and Jessica Hamerman², ¹Seattle Children's Hospital, Seattle, WA, ²Benaroya Research Institute, Seattle, WA, ³Feinstein Institutes for Medical Research, Manhasset, NY, ⁴Northwell Health, Cohen Children's Medical Center, New Hyde Park, NY, ⁵Seattle Children's Hospital, Seattle, WA, ⁶Cohen Children's Medical Center, Northwell Health, Lake Success, New York; Center for Autoimmune, Musculoskeletal, and Hematopoietic Diseases Research, Feinstein Institutes for Medical Research, Northwell Health, Manhasset, NY, ⁷Center for Autoimmune, Musculoskeletal, and Hematopoietic Diseases Research, Feinstein Institutes for Medical Research, Northwell Health, Manhasset, NY
Background/Purpose: Macrophage activation syndrome (MAS), a form of secondary hemophagocytic lymphohistiocytosis (HLH), is a potentially fatal complication of rheumatic diseases. MAS is a dysfunctional hyperinflammatory…
Abstract Number: 038 • 2023 Pediatric Rheumatology Symposium
HLA-DRB1*15 Alleles in Systemic Juvenile Idiopathic Arthritis with Lung Disease and Macrophage Activation Syndrome in Italy
Claudia Bracaglia¹, Manuela Pardeo¹, Maria Troiano², Giuseppe Testa², Ivan Caiello¹, Arianna De Matteis³, Matteo Trevisan¹, Franco Locatelli⁴, Marco Andreani² and Fabrizio De Benedetti¹, ¹Division of Rheumatology, IRCCS Ospedale Pediatrico Bambino Gesù, Roma, Italy, ²Laboratory of Transplant Immunogenetics, Department of Haematology/Oncology, Cell and Gene, IRCCS Ospedale Pediatrico Bambino Gesù, Roma, Lazio, Italy, ³Division of Rheumatology, IRCCS Ospedale Pediatrico Bambino Gesu', Roma, Italy, ⁴Ospedale Pediatrico Bambino Gesù, Department of Hematology/Oncology, Cell and Gene Therapy, Roma, Italy
Background/Purpose: Systemic juvenile idiopathic arthritis (sJIA) is characterized by unique clinical features and it is considered as a polygenic autoinflammatory disease. Macrophage activation syndrome (MAS)…
Abstract Number: 075 • 2023 Pediatric Rheumatology Symposium
Emapalumab Treatment Followed by Hematopoietic Stem Cell Transplantation in Systemic Juvenile Idiopathic Arthritis Complicated by Recurrent Macrophage Activation Syndrome
Claudia Bracaglia¹, Manuela Pardeo¹, Giulia Marucci², Simona Riccio², Francesco Quagliarella³, Ivan Caiello², Giusi Prencipe², Pietro Merli³, Franco Locatelli³ and Fabrizio De Benedetti¹, ¹Division of Rheumatology, IRCCS Ospedale Pediatrico Bambino Gesù, Roma, Italy, ²Division of Rheumatology, IRCCS Ospedale Pediatrico Bambino Gesu', Roma, Italy, ³Ospedale Pediatrico Bambino Gesù, Department of Hematology/Oncology, Cell and Gene Therapy, Roma, Italy
Background/Purpose: Macrophage activation syndrome (MAS) is a life-threatening complication of different rheumatic diseases, particularly of systemic juvenile idiopathic arthritis (sJIA).Methods: We report the case of…
Abstract Number: 079 • 2023 Pediatric Rheumatology Symposium
Dynamics of Neutrophil Activation in Repeated TLR-9-Induced Mouse Model of Macrophage Activation Syndrome
Natsumi Inoue, Richard Chhaing, Sanjeev Dhakal, Thuy Do and Grant Schulert, Cincinnati Children's Hospital Medical Center, Cincinnati, OH
Background/Purpose: Macrophage activation syndrome (MAS) is a life-threatening complication of rheumatic diseases including systemic juvenile idiopathic arthritis (SJIA). SJIA shows prominent neutrophil activation with expansion…
Abstract Number: 095 • 2023 Pediatric Rheumatology Symposium
Systemic Juvenile Idiopathic Arthritis Associated Lung Disease in Europe
Claudia Bracaglia¹, Francesca Minoia², Christoph Kessel³, Sebastiaan Vastert⁴, Manuela Pardeo¹, Alessia Arduini¹, Sarka Fingerhutova⁵, Irina Nikishina⁶, Ozge Basaran⁷, Nural Kiper⁸, Mikhail Kostik⁹, Mia Glerup¹⁰, Roberta Caorsi¹¹, AnnaCarin Horne¹², Giovanni Filocamo², Helmut Wittkowski³, Marija Jelusic¹³, Jordi Anton¹⁴, Samira Khaldi-Plassart¹⁵, Alexandre Belot¹⁵, Gerd Horneff¹⁶, Seraina Palmer Sarrott¹⁷, Elvira Cannizzaro Schneider¹⁸, Lampros Fotis¹⁹, Pavla Dolezalova⁵, Angelo Ravelli²⁰, Seza Ozen⁷ and Fabrizio De Benedetti¹, ¹Division of Rheumatology, IRCCS Ospedale Pediatrico Bambino Gesù, Roma, Italy, ²Fondazione IRCCS Ca' Grande Ospedale Maggiore Policlinico, Milano, Italy, ³Department of Pediatric Rheumatology & Immunology, WWU Medical Center (UKM), Muenster, Germany, ⁴Wilhelmina Children’s Hospital, Department of Pediatric Immunology and Rheumatology, Utrecht, The Netherlands, Utrecht, Netherlands, ⁵Centre 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, ⁶V.A. Nasonova Research Institute of Rheumatology, Moscow, Russia, ⁷Department of Pediatrics, Division of Pediatric Rheumatology, Hacettepe University, Ankara, Turkey, ⁸Department of Pediatrics, Division of Pediatric Pulmonology, Hacettepe University, Ankara, Turkey, ⁹Saint-Petersburg State Pediatric Medical University, Saint-Petersburg, Russia, ¹⁰Department of Pediatrics, Aarhus University Hospital, Aarhus, Denmark, ¹¹Department of Pediatrics and Rheumatology, IRRCS Istituto G. Gaslini, Genova, Italy, ¹²Department of pediatric rheumathology Karolinska University Hospital and Department of pediatrics, Karolinska Institute, Stockholm, Sweden, ¹³Department of Paediatrics, University of Zagreb School of Medicine, University Hospital Centre, 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, ¹⁶Pediatrics, Asklepios Clinic Sankt Augustin, Sankt Augustin, Germany, ¹⁷Paediatric Rheumatology University Children’s Hospital Zurich, Zurich, Zurich, Switzerland, ¹⁸Paediatric Rheumatology University Children’s Hospital Zurich, Zurich, Switzerland, ¹⁹Pediatric Rheumatology Division, 3rd Department of Pediatrics, National and Kapodistrian University of Athens,‘’ATTIKON’’ General University Hospital, Athens, Greece, ²⁰IRRCS Istituto Giannina Gaslini and Università degli Studi di Genova, Genova, Italy
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: 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…

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