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Pathogenesis and Treatment of Refractory Disease Courses in Systemic Juvenile Idiopathic Arthritis Refractory Arthritis, Recurrent Macrophage Activation Syndrome and Chronic Lung Disease

RHEUMATIC DISEASE CLINICS OF NORTH AMERICA (2021)

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RHEUMATIC DISEASE CLINICS OF NORTH AMERICA

卷 47, 期 4, 页码 585-606

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W B SAUNDERS CO-ELSEVIER INC

DOI: 10.1016/j.rdc.2021.06.003

关键词

Systemic JIA; Refractory disease; Translational research; Lung disease; Macrophage activation syndrome

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Rheumatology

资金

National Institutes of Arthritis and Musculoskeletal and Skin Diseases, National Institutes of Health [K08-AR072075]

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Tramper-Stranders, Andrew J. Pollard, Rama Kandasamy, Stephane Paulus, Michael J. Carter, Daniel O'Connor, Sagida Bibi, Dominic F. Kelly, Meeru Gurung, Stephen Thorson, Imran Ansari, David R. Murdoch, Shrijana Shrestha, Marieke Emonts, Emma Lim, Lucille Valentine, Karen Allen, Kathryn Bell, Adora Chan, Stephen Crulley, Kirsty Devine, Daniel Fabian, Sharon King, Paul McAlinden, Sam McDonald, Anne McDonnell, Ailsa Pickering, Evelyn Thomson, Amanda Wood, Diane Wallia, Phil Woodsford, Frances Baxter, Ashley Bell, Mathew Rhodes, Rachel Agbeko, Christine Mackerness, Bryan Baas, Lieke Kloosterhuis, Wilma Oosthoek, Tasnim Arif, Joshua Bennet, Kalvin Collings, Ilona van der Giessen, Alex Martin, Aqeela Rashid, Emily Rowlands, Gabriella de Vries, Fabian van der Velden, Mike Martin, Ravi Mistry, Ulrich von Both, Laura Kolberg, Manuela Zwerenz, Judith Buschbeck, Christoph Bidlingmaier, Vera Binder, Katharina Danhauser, Nikolaus Haas, Matthias Griese, Tobias Feuchtinger, Julia Keil, Matthias Kappler, Eberhard Lurz, Georg Muench, Karl Reiter, Carola Schoen, Francois Mallet, Karen Brengel-Pesce, Alexandre Pachot, Marine Mommert, Marko Pokorn, Mojca Kolnik, Katarina Vincek, Tina Plankar Srovin, Natalija Bahovec, Petra Prunk, Veronika Osterman, Tanja Avramoska, Taco Kuijpers, Ilse Jongerius, J. M. van den Berg, D. Schonenberg, A. M. Barendregt, D. Pajkrt, M. van der Kuip, A. M. van Furth, Evelien Sprenkeler, Judith Zandstra, G. van Mierlo, J. Geissler, Kenneth Baillie, Malcolm Gracie Semple, Gail Carson, Peter J. M. Openshaw, Jake Dunning, Laura Merson, Clark D. Russell, David Dorward, Maria Zambon, Meera Chand, Richard S. Tedder, Say Khoo, Lance C. W. Turtle, Tom Solomon, Samreen Ijaz, Tom Fletcher, Massimo Palmarini, Antonia Y. W. Ho, Emma Thomson, Nicholas Price, Judith Breuer, Thushan de Silva, Chloe Donohue, Hayley Hardwick, Wilna Oosthuyzen, Miranda Odam, Primrose Chikowore, Lauren Obosi, Sara Clohisey, Andrew Law, Lucy Norris, Sarah Tait, Murray Wham, Richard Clark, Audrey Coutts, Lorna Donelly, Angie Fawkes, Tammy Gilchrist, Katarzyna Hafezi, Louise MacGillivray, Alan Maclean, Sarah McCafferty, Kirstie Morrice, Lee Murphy, Nicola Wrobel, Sarah E. McDonald, Victoria Shaw, Jane A. Armstrong, Lauren Lett, Paul Henderson, Louisa Pollock, Shyla Kishore, Helen Brotherton, Lawrence Armstrong, Andrew Mita, Anna Dall, Kristyna Bohmova, Sheena Logan, Louise Gannon, Ken Agwuh, Srikanth Chukkambotla, Ingrid DuRand, Duncan Fullerton, Sanjeev Garg, Clive Graham, Tassos Grammatikopoulos, Stuart Hartshorn, Luke Hodgson, Paul Jennings, George Koshy, Tamas Leiner, James Limb, Jeff Little, Elijah Matovu, Fiona McGill, Craig Morris, John Morrice, David Price, Henrik Reschreiter, Tim Reynolds, Paul Whittaker, Rachel Tayler, Clare Irving, Maxine Ramsay, Margaret Millar, Barry Milligan, Naomy Hickey, Maggie Connon, Catriona Ward, Laura Beveridge, Susan MacFarlane, Karen Leitch, Claire Bell, Lauren Finlayson, Joy Dawson, Janie Candlish, Laura McGenily, Tara Roome, Cynthia Diaba, Jasmine Player, Natassia Powell, Ruth Howman, Sara Burling, Sharon Floyd, Sarah Farmer, Susie Ferguson, Susan Hope, Lucy Rubick, Rachel Swingler, Emma Collins, Collette Spencer, Amaryl Jones, Barbara Wilson, Diane Armstrong, Mark Birt, Holly Dickinson, Rosemary Harper, Darran Martin, Amy Roff, Sarah Mills

Summary: Over 1,000 cases of unexplained paediatric hepatitis have been reported worldwide, with 278 cases in the UK. A study investigated 38 cases and found high levels of adeno-associated virus 2 (AAV2) in the liver, blood, plasma or stool of 27 out of 28 cases. The study suggests that abnormal AAV2 replication aided by other viruses may trigger immune-mediated liver disease in predisposed children.

NATURE (2023)

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Article Multidisciplinary Sciences

Adeno-associated virus 2 infection in children with non-A-E hepatitis

Antonia Ho, Richard Orton, Rachel Tayler, Patawee Asamaphan, Vanessa Herder, Chris Davis, Lily Tong, Katherine Smollett, Maria Manali, Jay Allan, Konrad Rawlik, Sarah E. McDonald, Elen Vink, Louisa Pollock, Louise Gannon, Clair Evans, Jim McMenamin, Kirsty Roy, Kimberly Marsh, Titus Divala, Matthew T. G. Holden, Michael Lockhart, David Yirrell, Sandra Currie, Maureen O'Leary, David Henderson, Samantha J. Shepherd, Celia Jackson, Rory Gunson, Alasdair MacLean, Neil McInnes, Amanda Bradley-Stewart, Richard Battle, Jill A. Hollenbach, Paul Henderson, Miranda Odam, Primrose Chikowore, Wilna Oosthuyzen, Meera Chand, Melissa Shea Hamilton, Diego Estrada-Rivadeneyra, Michael Levin, Nikos Avramidis, Erola Pairo-Castineira, Veronique Vitart, Craig Wilkie, Massimo Palmarini, Surajit Ray, David L. Robertson, Ana da Silva Filipe, Brian J. Willett, Judith Breuer, Malcolm G. Semple, David Turner, J. Kenneth Baillie, Emma C. Thomson

Summary: An investigation found a possible association between AAV2 infection and host genetics in a recent outbreak of acute hepatitis in children in Scotland. The study used various methods to detect AAV2 infection in plasma and liver samples, and identified pathological features related to the virus in liver biopsy samples.

NATURE (2023)

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Article Immunology

Meningococcal ACWY conjugate vaccine immunogenicity and safety in adolescents with juvenile idiopathic arthritis and inflammatory bowel disease: A prospective observational cohort study

Milou Ohm, Joeri W. van Straalen, Marieke Zijlstra, Gerrie de Joode-Smink, Anne Jasmijn Sellies, Joost F. Swart, Sebastiaan J. Vastert, Joris M. van Montfrans, Marije Bartels, Annet van Royen-Kerkhof, Joanne G. Wildenbeest, Caroline A. Lindemans, Victorien M. Wolters, Roos A. W. Wennink, Joke H. de Boer, Mirjam J. Knol, Marloes W. Heijstek, Elisabeth A. M. Sanders, Frans M. Verduyn-Lunel, Guy A. M. Berbers, Nico M. Wulffraat, Marc H. A. Jansen

Summary: This study investigated the immunogenicity of the meningococcal serogroup ACWY conjugate vaccine in minors with juvenile idiopathic arthritis (JIA) and inflammatory bowel disease (IBD). The results showed that the concentrations of antibodies for serotypes A and W were lower in patients compared to healthy controls, and patients using anti-TNF therapy had lower immune response. Therefore, an extra booster vaccination should be considered for these patients.

VACCINE (2023)

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Article Cell Biology

Pyruvate metabolism controls chromatin remodeling during CD4+T cell activation

Enric Mocholi, Laura Russo, Keshav Gopal, Andrew G. Ramstead, Sophia M. Hochrein, Harmjan R. Vos, Geert Geeven, Adeolu O. Adegoke, Anna Hoekstra, Robert M. van Es, Jose Ramos Pittol, Sebastian Vastert, Jared Rutter, Timothy Radstake, Jorg van Loosdregt, Celia Berkers, Michal Mokry, Colin C. Anderson, Ryan M. O'Connell, Martin Vaeth, John Ussher, Boudewijn M. T. Burgering, Paul J. Coffer

Summary: Upon antigen-specific TCR engagement, the generation of extramitochondrial pyruvate is crucial for acetyl-CoA production and subsequent histone acetylation remodeling. PDH-deficient T cells show that PDH-dependent acetyl-CoA production is a rate-limiting step during T cell activation. This study highlights the integration of metabolic and histone-modifying enzymes in CD4+ T cell activation, suggesting a potential therapeutic approach for regulating antigen-driven T cell activation.

CELL REPORTS (2023)

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Review Pediatrics

The protective effect of COVID-19 vaccines on developing multisystem inflammatory syndrome in children (MIS-C): a systematic literature review and meta-analysis

Mohamad Hamad Saied, Laura van der Griend, Joeri W. van Straalen, Nico M. Wulffraat, Sebastiaan Vastert, Marc H. A. Jansen

Summary: This study conducted a systematic review and meta-analysis to investigate the effectiveness of current COVID-19 vaccines in preventing multisystem inflammatory syndrome in children (MIS-C) and adolescents. The findings suggest that mRNA COVID-19 vaccines in children aged 12-18 years are safe and significantly reduce the development of MIS-C. The risk of MIS-C post-vaccination is much lower compared to the risk of MIS-C post-infection.

PEDIATRIC RHEUMATOLOGY (2023)

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Article Pediatrics

Withdrawing biologics in non-systemic JIA: what matters to pediatric rheumatologists?

Janine van Til, Michelle M. A. Kip, Ellen J. H. Schatorje, Gillian Currie, Marinka M. Twilt, Susanne F. Benseler, Joost J. Swart, Sebastiaan Vastert, Nico Wulffraat, Rae S. M. Yeung, C. G. M. (Karin) Groothuis-Oudshoorn, Sanne Warta, Deborah A. Marshall, Maarten J. IJzerman

Summary: This study surveyed pediatric rheumatologists in Canada and the Netherlands to explore their views on withdrawing biologic therapy in children with clinically inactive non-systemic JIA and when they decide to postpone the withdrawal. The results showed that the strongest driver of the decision to postpone biologic therapy withdrawal was the preference of the child and/or parents, followed by flare in the current treatment period and uveitis in the current treatment period. On average, biologic therapy withdrawal was initiated 6.7 months later when the child or parent preferred to continue treatment.

PEDIATRIC RHEUMATOLOGY (2023)

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Article Rheumatology

The 2022 EULAR/ACR Points to Consider at the Early Stages of Diagnosis and Management of Suspected Haemophagocytic Lymphohistiocytosis/Macrophage Activation Syndrome (HLH/MAS)

Bita Shakoory, Ashley Geerlinks, Marta Wilejto, Kate Kernan, Melissa Hines, Micol Romano, David Piskin, Angelo Ravelli, Rashmi Sinha, Daniel Aletaha, Carl Allen, Hamid Bassiri, Edward M. Behrens, Joseph Carcillo, Linda Carl, Winn Chatham, Jeffrey I. Cohen, Randy Q. Cron, Erik Drewniak, Alexei A. Grom, Lauren A. Henderson, Annacarin Horne, Michael B. Jordan, Kim E. Nichols, Grant Schulert, Sebastiaan Vastert, Erkan Demirkaya, Raphaela Goldbach-Mansky, Fabrizio de Benedetti, Rebecca A. Marsh, Scott W. Canna

Summary: This study aims to provide evidence-based and consensus-based guidance for clinicians in the early diagnosis, treatment, and monitoring of HLH/MAS. Through research, questionnaires, and expert opinions, 6 overarching statements and 24 specific points to consider were developed, covering various aspects including early recognition, diagnosis, treatment, and monitoring. The involvement of multidisciplinary expertise is crucial for timely intervention and management.

ARTHRITIS & RHEUMATOLOGY (2023)

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Article Rheumatology

Recombinant Interleukin-1 Receptor Antagonist Is an Effective First-Line Treatment Strategy in New-Onset Systemic Juvenile Idiopathic Arthritis, Irrespective of HLA-DRB1 Background and IL1RN Variants

Remco G. A. Erkens, Jorg J. A. Calis, Anouk Verwoerd, Sytze De Roock, Nienke M. Ter Haar, Gerda Den Engelsman, Lars T. van der Veken, Robert F. Ernst, Hanneke W. M. Van Deutekom, Alex Pickering, Rianne C. Scholman, Marc H. A. Jansen, Joost F. Swart, Rashmi Sinha, Johannes Roth, Grant S. Schulert, Alexei A. Grom, Jorg Van Loosdregt, Sebastiaan J. Vastert

Summary: Using anakinra as first-line treatment in new-onset sJIA patients resulted in high rates of clinical inactive disease, regardless of HLA-DRB1 or IL1RN variants. Early initiation of biologic therapy may be more appropriate for patients with different genetic susceptibility backgrounds.

ARTHRITIS & RHEUMATOLOGY (2023)

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Article Rheumatology

Managing juvenile idiopathic arthritis within the context of their life: What we learnt from children and youth living with juvenile idiopathic arthritis and their parents

G. R. Currie, B. L. Kennedy, S. M. Benseler, R. S. M. Yeung, J. F. Swart, S. J. Vastert, N. M. Wulffraat, M. M. A. Kip, Gail MacKean, D. A. Marshall

Summary: This study aimed to explore the management experience of youth with Juvenile Idiopathic Arthritis (JIA) and their parents. Through semi-structured interviews with youth receiving biological medication for JIA and their parents, four themes were identified: aspects of life affected by JIA and its management, lived experience with JIA management, medication decision-making, and involvement in decision-making. The findings highlight the importance of person- and family-centred care in pediatric rheumatology.

MUSCULOSKELETAL CARE (2023)

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Article Rheumatology

J. Claude Bennett, MD: Scholar, Physician, and Leader

S. Louis Bridges, Steffen Gay

RHEUMATIC DISEASE CLINICS OF NORTH AMERICA (2024)

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Article Rheumatology

H. Ralph Schumacher

Joshua F. Baker, Daniel G. Baker

RHEUMATIC DISEASE CLINICS OF NORTH AMERICA (2024)

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Article Rheumatology

Hal Holman of Stanford

William Neal Roberts Jr, Edward R. Lew, Matthew H. Liang

RHEUMATIC DISEASE CLINICS OF NORTH AMERICA (2024)

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Article Rheumatology

Nathan Zvaifler

Daniel A. Albert

RHEUMATIC DISEASE CLINICS OF NORTH AMERICA (2024)

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Article Rheumatology

Gerald P Rodnan

Virginia D. Steen, Thomas A. Medsger

RHEUMATIC DISEASE CLINICS OF NORTH AMERICA (2024)

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Article Rheumatology

Eric Bywaters and Barbara Ansell Founders of Modern Pediatric Rheumatology

Patricia Woo, Ross E. Petty

RHEUMATIC DISEASE CLINICS OF NORTH AMERICA (2024)

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Article Rheumatology

John R. Ward, MD: Pioneer- Clinical Trials in Rheumatology

H. James Williams, Grant W. Cannon, Daniel O. Clegg

RHEUMATIC DISEASE CLINICS OF NORTH AMERICA (2024)

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Biographical-Item Rheumatology

Edmund Lawrence Dubois (1923-1985)

Daniel J. Wallace

RHEUMATIC DISEASE CLINICS OF NORTH AMERICA (2024)

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Article Rheumatology

Daniel J McCarty, MD: A Giant of Rheumatology

Ann K. Rosenthal, Mary E. Csuka, Paul Halverson

Summary: Dr. McCarty is a giant in the field of rheumatology. His clinical and scientific discoveries have had a major impact and benefited many patients. He has been an exceptional mentor and role model, influencing our clinical practice styles, the importance we place on clinical observation, and our approach to scientific questions.

RHEUMATIC DISEASE CLINICS OF NORTH AMERICA (2024)

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Article Rheumatology

Naomi Rothfield, MD, MACR A Giant Gift in a Small Package

Arthur Weinstein

RHEUMATIC DISEASE CLINICS OF NORTH AMERICA (2024)

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Article Rheumatology

The Giants of Rheumatology at Johns Hopkins

Marc C. Hochberg

RHEUMATIC DISEASE CLINICS OF NORTH AMERICA (2024)

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Article Rheumatology

E Carwile LeRoy, MD

Richard M. Silver

RHEUMATIC DISEASE CLINICS OF NORTH AMERICA (2024)

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Article Rheumatology

Walter Bauer, Marian Wilkins Ropes, and the Massachusetts General Hospital

Anthony M. Reginato, Michelle A. Petri, Jonathan Kay

Summary: Bauer and Ropes were influential figures in the field of rheumatology, making significant contributions through clinical observation and laboratory research. They were also exceptional mentors and leaders in the field.

RHEUMATIC DISEASE CLINICS OF NORTH AMERICA (2024)

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Article Rheumatology

Charles L Christian Model Physician Scientist and Mentor

Mary K. Crow, Josef S. Smolen

RHEUMATIC DISEASE CLINICS OF NORTH AMERICA (2024)

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Article Rheumatology

Lessons from Carl M. Pearson 1919-1981

James S. Louie

RHEUMATIC DISEASE CLINICS OF NORTH AMERICA (2024)

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