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  1. Article: The Big Bad Wolf: Macrophage Activation Syndrome in Childhood-Onset Systemic Lupus Erythematosus.

    Schulert, Grant S

    The Journal of rheumatology

    2022  Volume 49, Issue 10, Page(s) 1082–1084

    MeSH term(s) Humans ; Macrophage Activation Syndrome/etiology ; Lupus Erythematosus, Systemic/complications ; Case-Control Studies
    Language English
    Publishing date 2022-09-01
    Publishing country Canada
    Document type Editorial ; Comment
    ZDB-ID 194928-7
    ISSN 1499-2752 ; 0315-162X
    ISSN (online) 1499-2752
    ISSN 0315-162X
    DOI 10.3899/jrheum.220780
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    Zs.A 1168: Show issues Location:
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  2. Article ; Online: The IL-18/IFNγ axis in systemic JIA and MAS-new answers, more questions.

    Schulert, Grant S

    Rheumatology (Oxford, England)

    2021  Volume 60, Issue 7, Page(s) 3045–3047

    MeSH term(s) Arthritis, Juvenile ; Humans ; Interleukin-18 ; Macrophage Activation Syndrome
    Chemical Substances Interleukin-18
    Language English
    Publishing date 2021-04-09
    Publishing country England
    Document type Editorial ; Research Support, N.I.H., Extramural ; Comment
    ZDB-ID 1464822-2
    ISSN 1462-0332 ; 1462-0324
    ISSN (online) 1462-0332
    ISSN 1462-0324
    DOI 10.1093/rheumatology/keab342
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    Zs.MO 497: Show issues

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  3. Article ; Online: Can tocilizumab calm the cytokine storm of COVID-19?

    Schulert, Grant S

    The Lancet Rheumatology

    2020  Volume 2, Issue 8, Page(s) e449–e451

    Keywords covid19
    Language English
    Publishing date 2020-06-24
    Publishing country England
    Document type Journal Article
    ISSN 2665-9913
    ISSN (online) 2665-9913
    DOI 10.1016/S2665-9913(20)30210-1
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  4. Article ; Online: Molecular Pathways in the Pathogenesis of Systemic Juvenile Idiopathic Arthritis.

    Schulert, Grant S / Kessel, Christoph

    Rheumatic diseases clinics of North America

    2023  Volume 49, Issue 4, Page(s) 895–911

    Abstract: Systemic juvenile idiopathic arthritis (sJIA) is a rare childhood chronic inflammatory disorder with risk for life-threatening complications including macrophage activation syndrome and lung disease. At onset, sJIA pathogenesis resembles that of the ... ...

    Abstract Systemic juvenile idiopathic arthritis (sJIA) is a rare childhood chronic inflammatory disorder with risk for life-threatening complications including macrophage activation syndrome and lung disease. At onset, sJIA pathogenesis resembles that of the autoinflammatory periodic fever syndromes with marked innate immune activation, expansion of neutrophils and monocytes, and high levels of interleukin-18. Here, we review the current conceptual understanding of sJIA pathogenesis with a focus on both innate and adaptive immune pathways. Finally, we consider how recent progress toward understanding the immunologic basis of sJIA may support new therapies for refractory disease courses.
    MeSH term(s) Humans ; Child ; Arthritis, Juvenile ; Macrophage Activation Syndrome/complications
    Language English
    Publishing date 2023-07-21
    Publishing country United States
    Document type Journal Article ; Review
    ZDB-ID 92118-x
    ISSN 1558-3163 ; 0889-857X
    ISSN (online) 1558-3163
    ISSN 0889-857X
    DOI 10.1016/j.rdc.2023.06.007
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  5. Article ; Online: Complications of complications: diagnosis and treatment of recurrent macrophage activation syndrome in a patient with well-controlled systemic juvenile idiopathic arthritis.

    Macaraeg, Marci / Schulert, Grant S

    RMD open

    2023  Volume 9, Issue 1

    Abstract: Macrophage activation syndrome (MAS) is a subtype of haemophagocytic lymphohistiocytosis (HLH), and a well-described complication of systemic juvenile idiopathic arthritis (SJIA), triggered by disease onset or flare, infection, or some medications. Here, ...

    Abstract Macrophage activation syndrome (MAS) is a subtype of haemophagocytic lymphohistiocytosis (HLH), and a well-described complication of systemic juvenile idiopathic arthritis (SJIA), triggered by disease onset or flare, infection, or some medications. Here, we report a 20-year-old man with previously well-controlled SJIA, who developed first time MAS after acute Epstein-Barr virus (EBV) infection, with MAS recurrence due to a drug reaction, '3-week sulfasalazine syndrome', secondary to prophylactic trimethoprim/sulfamethoxazole. Both episodes of MAS were minimally responsive to pulse corticosteroids. Initial EBV-driven MAS was treated with multiple doses of emapalumab prior to resolution, while MAS secondary to sulfasalazine-induced 3-week syndrome required the initiation of ruxolitinib. This case exhibits two rare but life-threatening causes of MAS/secondary HLH in a single patient and the difficulties in their diagnosis and management.
    MeSH term(s) Male ; Humans ; Young Adult ; Adult ; Macrophage Activation Syndrome/complications ; Epstein-Barr Virus Infections/complications ; Arthritis, Juvenile/complications ; Sulfasalazine ; Herpesvirus 4, Human ; Lymphohistiocytosis, Hemophagocytic/complications ; Lymphohistiocytosis, Hemophagocytic/diagnosis
    Chemical Substances Sulfasalazine (3XC8GUZ6CB)
    Language English
    Publishing date 2023-01-21
    Publishing country England
    Document type Case Reports ; Journal Article
    ZDB-ID 2812592-7
    ISSN 2056-5933 ; 2056-5933
    ISSN (online) 2056-5933
    ISSN 2056-5933
    DOI 10.1136/rmdopen-2022-002611
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  6. Article ; Online: Mouse models of systemic juvenile idiopathic arthritis and macrophage activation syndrome.

    Inoue, Natsumi / Schulert, Grant S

    Arthritis research & therapy

    2023  Volume 25, Issue 1, Page(s) 48

    Abstract: Macrophage activation syndrome (MAS) is a life-threatening complication of pediatric rheumatic diseases, occurring most commonly in children with systemic juvenile idiopathic arthritis (SJIA). Despite several classes of currently available treatment ... ...

    Abstract Macrophage activation syndrome (MAS) is a life-threatening complication of pediatric rheumatic diseases, occurring most commonly in children with systemic juvenile idiopathic arthritis (SJIA). Despite several classes of currently available treatment options for SJIA, including biologic agents targeting IL-1 or IL-6, there remain severe cases suffering from refractory disease and recurrent MAS. The phenotype of MAS is similar to hemophagocytic lymphohistiocytosis (HLH), but the underlying pathophysiology of MAS complicating SJIA or other disorders has not been fully clarified. These facts make it challenging to develop and utilize animal models to study MAS. To date, there is no "perfect" model replicating MAS, but several models do demonstrate aspects of SJIA and/or MAS. In this review, we examine the proposed animal models of SJIA and MAS, focusing on how they reflect these disorders, what we have learned from the models, and potential future research questions. As we better understand the key features of each, animal models can be powerful tools to further define the pathophysiology of SJIA and MAS, and develop new treatment targets and strategies.
    MeSH term(s) Animals ; Mice ; Macrophage Activation Syndrome/complications ; Arthritis, Juvenile/complications ; Lymphohistiocytosis, Hemophagocytic/complications ; Biological Factors ; Disease Models, Animal
    Chemical Substances Biological Factors
    Language English
    Publishing date 2023-03-25
    Publishing country England
    Document type Journal Article ; Review ; Research Support, N.I.H., Extramural
    ZDB-ID 2107602-9
    ISSN 1478-6362 ; 1478-6354
    ISSN (online) 1478-6362
    ISSN 1478-6354
    DOI 10.1186/s13075-023-03032-8
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    Zs.A 5490: Show issues Location:
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  7. Article: The Storm Beneath the Storm: MAS-HLH in Inflammatory Myopathies.

    Schulert, Grant S

    The Journal of rheumatology

    2020  Volume 47, Issue 10, Page(s) 1461–1463

    MeSH term(s) Adult ; Humans ; Lymphohistiocytosis, Hemophagocytic ; Macrophage Activation Syndrome ; Myositis/drug therapy ; Prevalence ; Risk Factors
    Language English
    Publishing date 2020-10-01
    Publishing country Canada
    Document type Editorial ; Research Support, N.I.H., Extramural ; Comment
    ZDB-ID 194928-7
    ISSN 1499-2752 ; 0315-162X
    ISSN (online) 1499-2752
    ISSN 0315-162X
    DOI 10.3899/jrheum.191274
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  8. Article ; Online: New discoveries in the genetics and genomics of systemic juvenile idiopathic arthritis.

    Correia Marques, Mariana / Ombrello, Michael J / Schulert, Grant S

    Expert review of clinical immunology

    2024  , Page(s) 1–12

    Abstract: Introduction: Systemic juvenile idiopathic arthritis (sJIA) is a severe inflammatory condition with onset in childhood. It is sporadic, but elements of its stereotypical innate immune responses are likely genetically encoded by both common variants with ...

    Abstract Introduction: Systemic juvenile idiopathic arthritis (sJIA) is a severe inflammatory condition with onset in childhood. It is sporadic, but elements of its stereotypical innate immune responses are likely genetically encoded by both common variants with small effect sizes and rare variants with larger effects.
    Areas covered: Genomic investigations have defined the unique genetic architecture of sJIA. Identification of the class II HLA locus as the strongest sJIA risk factor for the first time brought attention to T lymphocytes and adaptive immune mechanisms in sJIA. The importance of the human leukocyte antigen (HLA) locus was reinforced by recognition that HLA-DRB1*15 alleles are strongly associated with development of drug reactions and sJIA-associated lung disease (sJIA-LD). At the
    Expert opinion: Genetic and genomic analysis of sJIA holds great promise for both basic discovery of the course and complications of sJIA, and may help guide personalized medicine and therapeutic decision-making.
    Language English
    Publishing date 2024-05-02
    Publishing country England
    Document type Journal Article ; Review
    ZDB-ID 2274260-8
    ISSN 1744-8409 ; 1744-666X
    ISSN (online) 1744-8409
    ISSN 1744-666X
    DOI 10.1080/1744666X.2024.2345868
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  9. Article ; Online: New developments related to lung complications in pediatric rheumatic disease.

    Rai, Shipra / Schulert, Grant S / Towe, Christopher

    Current opinion in rheumatology

    2023  Volume 35, Issue 5, Page(s) 273–277

    Abstract: Purpose of review: While substantial progress has been made understanding lung disease in adult patients with rheumatic disease, pediatric lung disease has not been well addressed. Several recent studies provide new insights into diagnosis, management ... ...

    Abstract Purpose of review: While substantial progress has been made understanding lung disease in adult patients with rheumatic disease, pediatric lung disease has not been well addressed. Several recent studies provide new insights into diagnosis, management and treatment of lung disease in children with rheumatic disease.
    Recent findings: Building on previous research, newly diagnosed patients may have abnormalities in pulmonary function tests and chest computed tomography imaging even when asymptomatic. New guidelines for screening for rheumatic-associated lung disease provide important recommendations for clinicians. New theories have been proposed about immunologic shifts leading to the development of lung disease in children with systemic juvenile idiopathic arthritis. Additionally, there are new antifibrotic agents that are being explored as treatments in pediatric patients with fibrotic lung diseases.
    Summary: Patients appear to have frequent lung function abnormalities while being clinically asymptomatic, emphasizing importance for rheumatologists to refer for pulmonary function tests and imaging at diagnosis. New advances are helping define optimal approaches to treatment of lung disease, including use of biologic agents and antifibrotic medicines for pediatric patients with rheumatologic diseases.
    MeSH term(s) Child ; Humans ; Rheumatic Diseases/complications ; Rheumatic Diseases/drug therapy ; Arthritis, Juvenile/complications ; Arthritis, Juvenile/drug therapy ; Arthritis, Juvenile/diagnosis ; Lung/diagnostic imaging ; Biological Factors ; Lung Diseases/etiology
    Chemical Substances Biological Factors
    Language English
    Publishing date 2023-05-04
    Publishing country United States
    Document type Review ; Journal Article ; Research Support, N.I.H., Extramural
    ZDB-ID 1045317-9
    ISSN 1531-6963 ; 1040-8711
    ISSN (online) 1531-6963
    ISSN 1040-8711
    DOI 10.1097/BOR.0000000000000947
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  10. Article ; Online: Can tocilizumab calm the cytokine storm of COVID-19?

    Schulert, Grant S

    The Lancet Rheumatology

    2020  Volume 2, Issue 8, Page(s) e449–e451

    Keywords covid19
    Language English
    Publisher Elsevier BV
    Publishing country us
    Document type Article ; Online
    ISSN 2665-9913
    DOI 10.1016/s2665-9913(20)30210-1
    Database BASE - Bielefeld Academic Search Engine (life sciences selection)

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