Cardiac involvement in adult-onset Still's disease: Manifestations, treatments and outcomes in a retrospective study of 28 patients

Bodard, Q.; Langlois, Vincent; Guilpain, Philippe; Quellec, A. Le; Vittecoq, Olivier; Noël, David; Eble, Vincent; Josse, S.; Schmidt, J.; Aouba, Achille; Lévesque, H.; Besnerais, M. Le; Benhamou, Y.

doi:10.1016/j.jaut.2020.102541

Cited by 33 publications

(39 citation statements)

References 39 publications

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“…Nevertheless, anakinra (an interleukin 1R antagonist) or tocilizumab can be initiated early for cortisone saving or if there is no improvement on corticosteroid therapy. Biotherapies can be immediately considered in the presence of severe systemic manifestations ( 6 ). In a recent study in 96 patients with AOSD, 28 (29%) presented cardiac involvement, 89% of these at diagnosis, represented mostly by pericarditis and rarely by cardiac tamponade, myocarditis, and 1 case of a noninfectious endocarditis.…”

Section: Discussionmentioning

confidence: 99%

“…In a recent study in 96 patients with AOSD, 28 (29%) presented cardiac involvement, 89% of these at diagnosis, represented mostly by pericarditis and rarely by cardiac tamponade, myocarditis, and 1 case of a noninfectious endocarditis. Patients with cardiac involvement were prone to having an intense inflammatory syndrome, having a refractory pattern to usual treatments, being more frequently admitted to the intensive care unit, and being more often treated with biotherapies ( 6 ).…”

Section: Discussionmentioning

confidence: 99%

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Acute Myocarditis Revealing Adult-Onset Still’s Disease

et al. 2021

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A 34-year-old man presented with fever, palpitations, maculopapular rash, pharyngitis, left cheilitis, and bilateral gonalgia. High-sensitivity troponin I concentration was 4,900 ng/l. Transthoracic echocardiogram revealed reduced global longitudinal strain. Cardiac magnetic resonance imaging showed acute myocarditis. Adult-onset Still’s disease was diagnosed, and treatment with intravenous corticosteroids and tocilizumab was initiated. ( Level of Difficulty: Beginner. )

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Section: Discussionmentioning

confidence: 99%

Section: Discussionmentioning

confidence: 99%

Acute Myocarditis Revealing Adult-Onset Still’s Disease

et al. 2021

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“…Moreover, the NETs' DNAbackbone has been demonstrated to enhance the activity of NE by protecting it from inhibition by antiproteases (39). Considering the pathogenic role of the interaction between neutrophils and endothelium in AOSD (40), NE-DNA complex may mediate vascular damage, leading to serositis and interstitial lung infiltration in AOSD. Further studies are needed to validate our findings and clarify the detailed role of NE-DNA in AOSD patients with cardiopulmonary manifestations.…”

Section: Discussionmentioning

confidence: 99%

Circulating Neutrophil Extracellular Traps Signature for Identifying Organ Involvement and Response to Glucocorticoid in Adult-Onset Still’s Disease: A Machine Learning Study

Jia

Wang

et al. 2020

Front. Immunol.

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“…AOSD is a rare disorder with potential cardiac involvement, serosal involvement occurs in 25%-60% of patients and might be severe, while pericarditis, myocarditis, cardiac tamponade are less prevalent. Endocardial involvement is rare, and can present as non-infective endocarditis (1,3,4). The IL-1 receptor antagonist (IL-1Rα) anakinra, which inhibits both IL-1α and IL-1β activity has been approved for the treatment of AOSD (2,5,6).…”

Section: Introductionmentioning

confidence: 99%

Autoinflammation Leading to Autoimmunity in Adult Onset Still’s Disease, More Than Simple Coincidence?

Valor‐Méndez

Manger

Cavallaro

et al. 2021

Preprint

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Background: Adult onset Still’s disease (AOSD) should be considered in the differential diagnosis of patients with endocarditis, with or without a cardiac decompensation. Case presentation: We report the case of a 68-year-old Caucasian male diagnosed with AOSD after an initial acute manifestation of endocarditis with severe aortic acute manifestation of endocarditis with severe aortic insufficiency. The histological findings revealed Libman-Sacks endocarditis. He was treated with the IL-1 receptor inhibitor anakinra. Two years later the patient developed a symptomatic dilated cardiomyopathy with reduced ejection fraction (23.5%) and functional anti-beta-1-adrenergic receptor antibodies, which was initially treated with plasmapheresis; anakinra was maintained. While his AOSD symptoms responded well, our patient presented with recurrent arthritis in multiple joints, dual energy-CT showed urate deposition compatible with a gouty arthropathy. Over seven years, he presented with recurrent episodes of arthritis and the adjustment of dosages of colchicine and febuxostat was needed. In 2018, our patient died due to a deterioration of his underlying cardiac disease. Conclusions: Only two cases with initial endocarditis prior to AOSD diagnosis have been published and we are not aware of any other cases reporting -β1AR-Ab development with DCM and gout in the setting of AOSD treated with anakinra.

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Cardiac involvement in adult-onset Still's disease: Manifestations, treatments and outcomes in a retrospective study of 28 patients

Cited by 33 publications

References 39 publications

Acute Myocarditis Revealing Adult-Onset Still’s Disease

Acute Myocarditis Revealing Adult-Onset Still’s Disease

Circulating Neutrophil Extracellular Traps Signature for Identifying Organ Involvement and Response to Glucocorticoid in Adult-Onset Still’s Disease: A Machine Learning Study

Autoinflammation Leading to Autoimmunity in Adult Onset Still’s Disease, More Than Simple Coincidence?

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