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Book ; Online: IL-1 Inhibition

Oliviero, Francesca / Sfriso, Paolo / Punzi, Leonardo / Dayer, Jean-Michel

2019

Keywords	Science: general issues ; Pharmacology ; Interleukin-1 ; Interleukin-1 receptor antagonist ; anti-interleukin-1 monoclonal antibody ; Inflammasomes ; Chronic inflammatory diseases ; Autoinflammatory diseases ; Arthritis ; Osteoathritis ; Skin Diseases ; crystal-induced inflammation
Size	1 electronic resource (217 pages)
Publisher	Frontiers Media SA
Document type	Book ; Online
Note	English ; Open Access
HBZ-ID	HT021230039
ISBN	9782889458578 ; 2889458571
Database	ZB MED Catalogue: Medicine, Health, Nutrition, Environment, Agriculture

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Article ; Online: Adult-Onset Still's Disease (AOSD): Advances in Understanding Pathophysiology, Genetics and Emerging Treatment Options.

Bindoli, Sara / Baggio, Chiara / Doria, Andrea / Sfriso, Paolo

Drugs

2024 Volume 84, Issue 3, Page(s) 257–274

Abstract: Adult-onset Still's disease (AOSD) is a multisystemic complex disorder clinically characterised by episodes of spiking fever, evanescent rash, polyarthritis or diffuse arthralgias; multiorgan involvement may develop according to the hyper-inflammatory ... ...

Abstract	Adult-onset Still's disease (AOSD) is a multisystemic complex disorder clinically characterised by episodes of spiking fever, evanescent rash, polyarthritis or diffuse arthralgias; multiorgan involvement may develop according to the hyper-inflammatory extent. The pathogenesis of AOSD is not completely recognised. The central role of macrophage activation, which results in T helper 1 (Th1) cell cytokine activation, is well established. Pro-inflammatory cytokines such as interleukin (IL)-1, IL-6 and IL-18 play a fundamental role in disease onset and progression. The disease may develop in both children and adults with overlapping clinical features, and although several subsets depending on the clinical manifestations and the cytokines expressed have been identified, the dichotomy between systemic juvenile idiopathic arthritis (sJIA) and AOSD nowadays has been overcome, and the pathology is considered a disease continuum between ages. Various therapeutic approaches have been evaluated thus far, and different compounds are under assessment for AOSD treatment. Historically, glucocorticoids have been employed for treating systemic manifestations of Still's disease, while conventional synthetic disease modifying anti-rheumatic drugs (csDMARDs) demonstrated efficacy in controlling the articular manifestations. Currently, biological (b) DMARDs are widely employed; IL-1 inhibitors such as anakinra and canakinumab have proven to have high efficacy and an excellent safety profile and the anti-IL-6 tocilizumab is approved for sJIA, with several trials and longitudinal studies confirming its efficacy and safety. Moreover, in the light of the 'window of opportunity', new evidence showed that the earlier these treatments are initiated, the sooner clinical inactivity can be achieved. Other treatment options are being considered since several molecules involved in the disease pathophysiology can be targeted through various mechanisms. This review will provide a broad overview of AOSD pathophysiology, insights into specific organ manifestations and the currently available treatments with the identification of potential therapeutic targets involved in AOSD pathogenesis will be outlined.
MeSH term(s)	Adult ; Child ; Humans ; Still's Disease, Adult-Onset/drug therapy ; Antirheumatic Agents/therapeutic use ; Cytokines ; Interleukin-1/therapeutic use ; Fever/drug therapy
Chemical Substances	Antirheumatic Agents ; Cytokines ; Interleukin-1
Language	English
Publishing date	2024-03-05
Publishing country	New Zealand
Document type	Journal Article ; Review
ZDB-ID	120316-2
ISSN	1179-1950 ; 0012-6667
ISSN (online)	1179-1950
ISSN	0012-6667
DOI	10.1007/s40265-024-01993-x
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Article ; Online: Pulmonary alveolar proteinosis in an adult patient affected by Still's disease and recurrent episodes of macrophage activation syndrome.

Bindoli, Sara / Lococo, Sara / Calabrese, Fiorella / Sfriso, Paolo

Joint bone spine

2023 Volume 91, Issue 1, Page(s) 105654

MeSH term(s)	Adult ; Humans ; Macrophage Activation Syndrome/complications ; Macrophage Activation Syndrome/diagnosis ; Pulmonary Alveolar Proteinosis/diagnostic imaging ; Pulmonary Alveolar Proteinosis/therapy ; Arthritis, Juvenile/drug therapy ; Antirheumatic Agents/therapeutic use ; Still's Disease, Adult-Onset/drug therapy
Chemical Substances	Antirheumatic Agents
Language	English
Publishing date	2023-10-17
Publishing country	France
Document type	Letter
ZDB-ID	2020487-5
ISSN	1778-7254 ; 1297-319X
ISSN (online)	1778-7254
ISSN	1297-319X
DOI	10.1016/j.jbspin.2023.105654
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Zs.A 4590: Show issues

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Article ; Online: Intravenous anakinra to curb cytokine storm in adult-onset Still's disease and in macrophage activation syndrome: A case series.

Bindoli, Sara / Galozzi, Paola / Doria, Andrea / Sfriso, Paolo

Joint bone spine

2023 Volume 90, Issue 2, Page(s) 105524

Abstract: Objective: Adult-onset Still's disease (AOSD) is an auto-inflammatory polygenic disorder, for which the diagnosis is essentially clinical. The exclusion of mimickers [such as common bacterial and viral infections, hematologic malignancies, and, more ... ...

Abstract	Objective: Adult-onset Still's disease (AOSD) is an auto-inflammatory polygenic disorder, for which the diagnosis is essentially clinical. The exclusion of mimickers [such as common bacterial and viral infections, hematologic malignancies, and, more recently, severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2)] is necessary to confirm the diagnosis. Anti-interleukin (IL)-1 therapy is considered a treatment milestone for AOSD. Herein, we present a short series of newly-diagnosed AOSD or upcoming macrophage activation syndrome (MAS) cases who received intravenous (IV) anakinra, an IL-1 receptor blocker. Methods: Four patients with newly-diagnosed AOSD or upcoming MAS were treated with IV anakinra at the Rheumatology Unit of Padova University Hospital, Italy. We obtained informed consent from the patients for use of their cases and medical images for publication purposes. Results: All patients presented with AOSD or MAS during the COVID-19 pandemic, making diagnosis challenging due to similar immunological and clinical characteristics across both pathologies. All patients presented with hyperpyrexia and elevated inflammatory markers; two patients had a skin rash typically seen in AOSD. IV anakinra slowed down AOSD progression in all patients, prevented severe outcomes and mitigated the risk of multiorgan failure. All cases improved within 24hours of anakinra administration. Conclusion: We found that administration of anakinra in patients with newly-diagnosed AOSD and/or upcoming MAS reduced hyperinflammation and prevented life-threatening complications. The IV route appears to be preferable in the hospital setting, where comorbidities such as coagulopathies and thrombocytopenia can complicate the use of other routes of administration.
MeSH term(s)	Adult ; Humans ; Interleukin 1 Receptor Antagonist Protein/therapeutic use ; Still's Disease, Adult-Onset/complications ; Still's Disease, Adult-Onset/diagnosis ; Still's Disease, Adult-Onset/drug therapy ; Macrophage Activation Syndrome/diagnosis ; Macrophage Activation Syndrome/drug therapy ; Macrophage Activation Syndrome/etiology ; Cytokine Release Syndrome/etiology ; Cytokine Release Syndrome/complications ; Pandemics ; COVID-19/complications ; SARS-CoV-2
Chemical Substances	Interleukin 1 Receptor Antagonist Protein
Language	English
Publishing date	2023-01-06
Publishing country	France
Document type	Case Reports
ZDB-ID	2020487-5
ISSN	1778-7254 ; 1297-319X
ISSN (online)	1778-7254
ISSN	1297-319X
DOI	10.1016/j.jbspin.2023.105524
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Article ; Online: JAK inhibitors for the treatment of VEXAS syndrome.

Bindoli, Sara / Baggio, Chiara / Doria, Andrea / Bertoldo, Eugenia / Sfriso, Paolo

Experimental biology and medicine (Maywood, N.J.)

2023 Volume 248, Issue 5, Page(s) 394–398

Abstract: Vacuoles, E1 enzyme, X-linked, autoinflammatory, somatic (VEXAS) syndrome is a novel described autoinflammatory entity for which the diagnosis is defined by somatic mutations of ... ...

Abstract	Vacuoles, E1 enzyme, X-linked, autoinflammatory, somatic (VEXAS) syndrome is a novel described autoinflammatory entity for which the diagnosis is defined by somatic mutations of the
MeSH term(s)	Male ; Humans ; Aged ; Janus Kinase Inhibitors/therapeutic use ; Genes, X-Linked ; Hematopoietic Stem Cells ; Myelodysplastic Syndromes ; Mutation
Chemical Substances	Janus Kinase Inhibitors
Language	English
Publishing date	2023-05-26
Publishing country	England
Document type	Case Reports ; Journal Article ; Review
ZDB-ID	4015-0
ISSN	1535-3699 ; 1525-1373 ; 0037-9727
ISSN (online)	1535-3699 ; 1525-1373
ISSN	0037-9727
DOI	10.1177/15353702231165030
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Article: Progress in Biological Therapies for Adult-Onset Still's Disease.

Galozzi, Paola / Bindoli, Sara / Doria, Andrea / Sfriso, Paolo

Biologics : targets & therapy

2022 Volume 16, Page(s) 21–34

Abstract: Adult-onset Still's disease (AOSD) is a rare multifactorial autoinflammatory disorder of unknown etiology, characterized by an excessive release of cytokines triggered by dysregulated inflammation and articular and systemic manifestations. The clinical ... ...

Abstract	Adult-onset Still's disease (AOSD) is a rare multifactorial autoinflammatory disorder of unknown etiology, characterized by an excessive release of cytokines triggered by dysregulated inflammation and articular and systemic manifestations. The clinical spectrum of AOSD ranges from self-limiting forms with mild symptoms to life-threatening cases and presents clinical and biological similarities with the juvenile form (sJIA). Nowadays, the advances in biologic agents no longer limit the treatment to NSAIDs, glucocorticoids, or conventional synthetic DMARDs. The blockade of IL-1 and IL-6 is effective in the treatment of systemic and articular inflammation of AOSD patients; however, novel compounds with different properties and targets are now available and others are being studied. In this review, starting from the pathogenesis of AOSD, we summarized the current and emerging biological therapies, possible effective agents for achieving AOSD control and remission.
Language	English
Publishing date	2022-04-21
Publishing country	New Zealand
Document type	Journal Article ; Review
ZDB-ID	2415708-9
ISSN	1177-5491 ; 1177-5475
ISSN (online)	1177-5491
ISSN	1177-5475
DOI	10.2147/BTT.S290329
Database	MEDical Literature Analysis and Retrieval System OnLINE

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Article ; Online: Anti-Inflammatory and Hypouricemic Effect of Bioactive Compounds: Molecular Evidence and Potential Application in the Management of Gout.

Scanu, Anna / Luisetto, Roberto / Ramonda, Roberta / Spinella, Paolo / Sfriso, Paolo / Galozzi, Paola / Oliviero, Francesca

Current issues in molecular biology

2022 Volume 44, Issue 11, Page(s) 5173–5190

Abstract: Gout is caused by the deposition of monosodium urate crystals in the joint and represents the most common form of inflammatory arthritis in men. Its prevalence is rising worldwide mainly due to the increase of risk factors associated with the disease, in ...

Abstract	Gout is caused by the deposition of monosodium urate crystals in the joint and represents the most common form of inflammatory arthritis in men. Its prevalence is rising worldwide mainly due to the increase of risk factors associated with the disease, in particular hyperuricemia. Besides gout, hyperuricemia leads to an increased inflammatory state of the body with consequent increased risk of comorbidities such as cardiovascular diseases. Increasing evidence shows that bioactive compounds have a significant role in fighting inflammatory and immune chronic conditions. In gout and hyperuricemia, these molecules can exert their effects at two levels. They can either decrease serum uric acid concentrations or fight inflammation associated with monosodium urate crystals deposits and hyperuricemia. In this view, they might be considered valuable support to the pharmacological therapy and prevention of the disease. This review aims to provide an overview of the beneficial role of bioactive compounds in hyperuricemia, gout development, and inflammatory pathways of the disease.
Language	English
Publishing date	2022-10-25
Publishing country	Switzerland
Document type	Journal Article ; Review
ZDB-ID	2000024-8
ISSN	1467-3045 ; 1467-3037
ISSN (online)	1467-3045
ISSN	1467-3037
DOI	10.3390/cimb44110352
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Article ; Online: IL-18 in Autoinflammatory Diseases: Focus on Adult Onset Still Disease and Macrophages Activation Syndrome.

Baggio, Chiara / Bindoli, Sara / Guidea, Irina / Doria, Andrea / Oliviero, Francesca / Sfriso, Paolo

International journal of molecular sciences

2023 Volume 24, Issue 13

Abstract: Interleukin-18 (IL-18) is a potent pro-inflammatory cytokine that is involved in various innate and adaptive immune processes related to infection, inflammation, and autoimmunity. Therefore, it is described as a key mediator of autoinflammatory diseases ... ...

Abstract	Interleukin-18 (IL-18) is a potent pro-inflammatory cytokine that is involved in various innate and adaptive immune processes related to infection, inflammation, and autoimmunity. Therefore, it is described as a key mediator of autoinflammatory diseases associated with the development of macrophage activation syndrome (MAS), including systemic juvenile idiopathic arthritis and adult-onset Still's disease. This review focuses on the role of IL-18 in inflammatory responses, placing emphasis on autoinflammatory diseases associated with chronic excess of serum IL-18, which correlate with clinical and biological signs of the disease. Therefore, it is useful for the diagnosis and monitoring of disease activity. Researchers are currently investigating IL-18's role as a therapeutic target for the treatment of inflammatory diseases. The inhibition of IL-18 signaling through recombinant human IL-18BP (IL-18 binding protein) seems to be an effective therapeutic strategy, though further studies are necessary to clarify its importance as a therapeutic target.
MeSH term(s)	Adult ; Humans ; Interleukin-18/metabolism ; Still's Disease, Adult-Onset/diagnosis ; Still's Disease, Adult-Onset/drug therapy ; Macrophage Activation Syndrome/diagnosis ; Hereditary Autoinflammatory Diseases ; Macrophages/metabolism
Chemical Substances	Interleukin-18
Language	English
Publishing date	2023-07-05
Publishing country	Switzerland
Document type	Journal Article ; Review
ZDB-ID	2019364-6
ISSN	1422-0067 ; 1422-0067 ; 1661-6596
ISSN (online)	1422-0067
ISSN	1422-0067 ; 1661-6596
DOI	10.3390/ijms241311125
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Article ; Online: The revisited role of interleukin-1 alpha and beta in autoimmune and inflammatory disorders and in comorbidities.

Galozzi, Paola / Bindoli, Sara / Doria, Andrea / Sfriso, Paolo

Autoimmunity reviews

2021 Volume 20, Issue 4, Page(s) 102785

Abstract: The interleukin (IL) 1 family of cytokines is noteworthy to have pleiotropic functions in inflammation and acquired immunity. Over the last decades, several progresses have been made in understanding the function and regulation of the prototypical ... ...

Abstract	The interleukin (IL) 1 family of cytokines is noteworthy to have pleiotropic functions in inflammation and acquired immunity. Over the last decades, several progresses have been made in understanding the function and regulation of the prototypical inflammatory cytokine (IL-1) in human diseases. IL-1α and IL-1β deregulated signaling causes devastating diseases manifested by severe acute or chronic inflammation. In this review, we examine and compare the key aspects of IL-1α and IL-1β biology and regulation and discuss their importance in the initiation and maintenance of inflammation that underlie the pathology of many human diseases. We also report the current and ongoing inhibitors of IL-1 signaling, targeting IL-1α, IL-1β, their receptor or other molecular compounds as effective strategies to prevent or treat the onset and progression of various inflammatory disorders.
MeSH term(s)	Adaptive Immunity ; Humans ; Inflammation ; Interleukin-1alpha ; Interleukin-1beta ; Signal Transduction
Chemical Substances	Interleukin-1alpha ; Interleukin-1beta
Language	English
Publishing date	2021-02-20
Publishing country	Netherlands
Document type	Journal Article ; Review
ZDB-ID	2144145-5
ISSN	1873-0183 ; 1568-9972
ISSN (online)	1873-0183
ISSN	1568-9972
DOI	10.1016/j.autrev.2021.102785
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Article ; Online: Hyperinflammation after anti-SARS-CoV-2 mRNA/DNA vaccines successfully treated with anakinra: Case series and literature review.

Bindoli, Sara / Giollo, Alessandro / Galozzi, Paola / Doria, Andrea / Sfriso, Paolo

Experimental biology and medicine (Maywood, N.J.)

2022 Volume 247, Issue 4, Page(s) 338–344

Abstract: The current SARS-CoV-2 pandemic diffused worldwide has encouraged the rapid development of vaccines to counter the spread of the virus. At present in Italy, 75.01% of the population completed the vaccination course (AIFA.gov.it) and very few adverse ... ...

Abstract	The current SARS-CoV-2 pandemic diffused worldwide has encouraged the rapid development of vaccines to counter the spread of the virus. At present in Italy, 75.01% of the population completed the vaccination course (AIFA.gov.it) and very few adverse events have been recorded by now. Side-effects related to a theoretical over-reaction of the immune system in response to vaccines administration have been described, and the possibility that an autoimmune or a hyperinflammatory condition may occur was recently observed. Herein, we report four cases of hyperinflammatory syndrome with features indicative of Adult-onset Still's disease (AOSD) and macrophage activation syndrome (MAS), occurred after anti-SARS-CoV-2 vaccine injection and seen at our Unit between March and May 2021. Since interleukin (IL)-1 is one of the pivotal cytokines involved in AOSD pathogenesis, the inhibition of IL-1 is crucial in ameliorating the clinical symptoms of those patients. Moreover, it has been highlighted the central role of IL-1 as a hallmark of the hyperinflammatory status elicited by SARS-CoV-2 infection. In this case series, we successfully employed the IL-1 receptor antagonist anakinra to curb the cytokine release likely unleashed by the vaccine stimulation in potentially predisposed subjects. We also made a literature search to detect other patients with hyperinflammation temporally related to vaccines injection who benefited from IL-1 inhibition, while other AOSD/MAS-like described syndromes improved with other immunomodulatory strategies.
MeSH term(s)	BNT162 Vaccine/adverse effects ; COVID-19 Vaccines/adverse effects ; ChAdOx1 nCoV-19/adverse effects ; Female ; Humans ; Inflammation/chemically induced ; Inflammation/drug therapy ; Interleukin 1 Receptor Antagonist Protein/therapeutic use ; Interleukin-1/immunology ; Interleukin-1/metabolism ; Male ; Middle Aged ; Still's Disease, Adult-Onset/chemically induced ; Still's Disease, Adult-Onset/etiology ; Vaccines, DNA/adverse effects ; mRNA Vaccines/adverse effects
Chemical Substances	COVID-19 Vaccines ; Interleukin 1 Receptor Antagonist Protein ; Interleukin-1 ; Vaccines, DNA ; mRNA Vaccines ; ChAdOx1 nCoV-19 (B5S3K2V0G8) ; BNT162 Vaccine (N38TVC63NU)
Language	English
Publishing date	2022-01-22
Publishing country	England
Document type	Case Reports ; Journal Article ; Review
ZDB-ID	4015-0
ISSN	1535-3699 ; 1525-1373 ; 0037-9727
ISSN (online)	1535-3699 ; 1525-1373
ISSN	0037-9727
DOI	10.1177/15353702211070290
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