Cited by 74Autoimmune and Inflammatory Disorders ResearchKawasaki Disease and Coronary ComplicationsRheumatoid Arthritis Research and Therapies

Adult-onset Still’s disease biological treatment strategy may depend on the phenotypic dichotomy

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Vercruysse, François, Barnetche, Thomas, Lazaro, Estibaliz, Shipley, Emilie, Lifermann, François, Balageas, Alexandre, Delbrel, Xavier, Fautrel, Bruno +3 more·2019·Arthritis Research & Therapy

Abstract

OBJECTIVES: Adult-onset Still's disease (AOSD) phenotype appears to be dichotomized in systemic or chronic articular forms. As biologicals and particularly interleukin (IL)-1 and IL-6 blockers play a more and more prominent role in the treatment, their place requires clarification. This study aimed to identify factors predictive of treatment response to anakinra or tocilizumab and investigate whether the choice of biotherapy and delays in the initiation of biotherapy influenced the likelihood of steroid discontinuation. METHODS: A multicenter exploratory retrospective study included all patients diagnosed with AOSD and receiving biological treatments in three regional hospitals until 2018. Clinical and biological characteristics at diagnosis and treatment-related data were collected. The nonparametric Mann-Whitney test was used to perform univariate analysis for quantitative variables, and Fisher's exact test was used for qualitative variables. RESULTS: Twenty-seven patients were included. All but one patient achieved remission with either anakinra or tocilizumab. Treatment responses depended on disease phenotype: the presence of arthritis and a chronic articular phenotype were associated with a substantial response to tocilizumab with p = 0.0009 (OR 36 [2.6-1703]) and p = 0.017 (OR 10 [1.22-92.6]), respectively, whereas the systemic form and the absence of arthritis were associated with a substantial response to anakinra with p = 0.0009 (OR 36 [2.6-1703]) and p = 0.017 (OR 10 [1.22-92.6]), respectively. Tocilizumab increased the likelihood of corticosteroid withdrawal (p = 0.029) regardless of delays in initiation or when it was initiated relative to other treatment in the overall therapeutic strategy. CONCLUSION: This study highlights the therapeutic implications of the phenotypic dichotomy of AOSD and should help us better codify AOSD treatment.

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  • Biomarkers effectively predict which patients with adult-onset Still's disease will have a response to anakinra or tocilizumab.

    “Likewise, biomarkers were recently shown to effectively predict which patients with adult-onset Still’s disease would have a response to anakinra or tocilizumab.111”

    From Cytokine storm (2020) · cites this paper as [111]

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Research works printed but not linked: 1

  1. [30]Gerfaud-Valentin M Maucort-Boulch D Hot A Iwaz J Ninet J Durieu I Adult-onset still disease: manifestations, treatment, outcome, and prognostic factors in 57 patients Medicine (Baltimore) 2014 93 2 91 99 24646465 PMC4616309

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Authors
Vercruysse, F., Barnetche, T., Lazaro, E., Shipley, E., Lifermann, F., Balageas, A., Delbrel, X., Fautrel, B., Richez, C., Schaeverbeke, T., & Truchetet, M.-E.
Affiliations
  • Centre Hospitalier Universitaire de Bordeaux (FR)
  • Centre Hospitalier de Dax –Côte d’Argent (FR)
  • Centre Hospitalier De Pau (FR)
  • Pitié-Salpêtrière Hospital (FR)
Corresponding author
Vercruysse, François
Venue
Arthritis Research & Therapy
Published
2019-02-12
Volume
21
Issue
1
Pages
53-53
ISSN
  • 1478-6354 (ISSN-L)
  • 1478-6362
OpenAlex
W2925874700
Cited by
74
References
53
Citations by year
2026: 3 · 2025: 4 · 2024: 8 · 2023: 9 · 2022: 18 · 2021: 14 · 2020: 9 · 2019: 8
FWCI
8.65
Type
journal article
Language
en
Open access
Locations
  • Arthritis Research & Therapy (publishedVersion, cc-by)
  • Europe PMC (PubMed Central) (publishedVersion, other-oa)
  • PubMed Central (publishedVersion, cc-by)
  • PubMed (publishedVersion)
  • HAL (submittedVersion, other-oa)
  • DOAJ (DOAJ: Directory of Open Access Journals) (submittedVersion, cc-by-sa)
Topics
  • Health Sciences › Medicine › Hematology › Autoimmune and Inflammatory Disorders Research
  • Health Sciences › Medicine › Surgery › Kawasaki Disease and Coronary Complications
  • Health Sciences › Medicine › Rheumatology › Rheumatoid Arthritis Research and Therapies
Keywords
adult-onset Still's disease, biologic therapy, anakinra, tocilizumab, disease phenotypes, corticosteroid withdrawal, treatment response prediction, remission
MeSH terms
Adult, Cartilage, Articular, Female, Hospitals, Community, Humans, Male, Middle Aged, Phenotype, Remission Induction, Retrospective Studies, Still's Disease, Adult-Onset, Outcome Assessment, Health Care, Antirheumatic Agents, Interleukin 1 Receptor Antagonist Protein, Young Adult, Antibodies, Monoclonal, Humanized

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