Treatment of refractory adult onset Still’s disease with tocilizumab—a single centre experience and literature review

Crnogaj, Mirna Reihl; Čubelić, Darija; Babić, Antonija; Mayer, Miroslav; Anić, Branimir

doi:10.1007/s00296-020-04622-4

Cited by 17 publications

(8 citation statements)

References 41 publications

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“…Similar to the previous studies, the most commonly preferred conventional immunosuppressive drug before the commencement of biological drug was methotrexate. [ 23 , 27 , 28 ] Cyclosporine was the second most common preferred drug for patients with hepatic transaminase elevations and who were receiving anakinra. Leflunomide was the drug secondly prescribed for the patients with mainly articular symptoms and who were receiving tocilizumab, consistent with the literature.…”

Section: Discussionmentioning

confidence: 99%

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Biological treatment in resistant adult-onset Still’s disease: A single-center, retrospective cohort study

et al. 2021

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Objectives: The aim of this study was to assess the demographic and clinical characteristics of patients with adult-onset Still’s disease (AOSD) under biological treatment. Patients and methods: This retrospective cohort study included a total of 19 AOSD patients (13 males, 6 females; median age: 37 years; range, 28 to 52 years) who received biological drugs due to refractory disease between January 2008 and January 2020. The data of the patients were obtained from the patient files. The response to the treatment was evaluated based on clinical and laboratory assessments at third and sixth follow-up visits. Results: Interleukin (IL)-1 inhibitor was prescribed for 13 (68.4%) patients and IL-6 inhibitor prescribed for six (31.6%) patients. Seventeen (89.5%) patients experienced clinical remission. Conclusion: Biological drugs seem to be effective for AOSD patients who are resistant to conventional therapies. Due to the administration methods and the high costs of these drugs, however, tapering the treatment should be considered, after remission is achieved.

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Section: Discussionmentioning

confidence: 99%

“…Frequently, lengthening the dosing interval or reducing the dose were preferred methods. [ 29 ] In the study presented by Reihl Crnogaj et al,[ 28 ] three of four patients had disease flares after cessation of tocilizumab due to the remission of the disease.…”

Section: Discussionmentioning

confidence: 99%

Biological treatment in resistant adult-onset Still’s disease: A single-center, retrospective cohort study

et al. 2021

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“… 89 In addition, there is increasing evidence of uncontrolled T-cell and macrophage activation – inducing proinflammatory cytokines, such as IL-1, IL-6, IL-18, TNF-α, and interferon (IFN)-γ overproduction – playing a crucial role in the pathogenesis of AOSD. 15 , 51 , 106 , 111 …”

Section: Adult Onset Still’s Diseasementioning

confidence: 99%

“…NSAIDs, disease-modifying anti-rheumatic drugs (DMARDs), glucocorticoids or corticosteroids have been used as conventional treatments for AOSD. 40 – 44 , 51 , 136 Tocilizumab, an IL-6 inhibitor, has shown efficacy in the treatment of patients with refractory AOSD. 50 – 52 , 137 , 138 Data from a multicenter retrospective study involving 27 patients ⩾ 16 years of age with AOSD showed that the remission was achieved in all patients, except one, with either anakinra or tocilizumab treatment.…”

Section: Adult Onset Still’s Diseasementioning

confidence: 99%

New insights on multigenic autoinflammatory diseases

Efthimiou¹,

Petryna

Nakasato

et al. 2022

Therapeutic Advances in Musculoskeletal

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Autoinflammatory diseases are disorders of the innate immune system, which can be either monogenic due to a specific genetic mutation or complex multigenic due to the involvement of multiple genes. The aim of this review is to explore and summarize the recent advances in pathogenesis, diagnosis, and management of genetically complex autoinflammatory diseases, such as Schnitzler’s syndrome; adult-onset Still’s disease; synovitis, acne, pustulosis, hyperostosis, osteitis syndrome/chronic recurrent multifocal osteomyelitis/chronic non-bacterial osteomyelitis; Adamantiades-Behçet’s disease; Yao syndrome; and periodic fever with aphthous stomatitis, pharyngitis, and adenitis syndrome. The PubMed database was screened for relevant articles using free text words and specific search strings. The search was limited to English-language articles, reporting the results of studies in humans, published through March 2021. Evidence from literature suggest that these rare multigenic autoinflammatory diseases can present with different clinical features and the diagnosis of these diseases can be challenging due to a combination of nonspecific manifestations that can be seen in a variety of other conditions. Diagnostic delays and disease complications may occur due to low disease awareness and the lack of pathognomonic markers. The pathogeneses of these diseases are complex and in some cases precise pathogenesis is not clearly understood. Conventional treatments are commonly used for the management of these conditions, but biologics have shown promising results. Biologics targeting proinflammatory cytokines including IL-1, IL-6, TNF-α, IL-17A and IL-18 have been shown to ameliorate signs and symptoms of different multigenic autoinflammatory diseases.

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“…The age distribution of the AOSD is bimodal, with one peak occurring between 15-25 years and the other between 36-45 years ( 5 ). The typical clinical manifestations of AOSD mainly include fever (60-100%), arthritis or arthralgia (70-100%) and maculopapular red rash (60-80%) ( 6 ). Atypical clinical manifestations such as sore throat or pharyngitis, myalgia, lymphadenopathy, and hepatosplenomegaly may also occur in some patients ( 7 ).…”

Section: Introductionmentioning

confidence: 99%

Bibliometrics analysis on the research status and trends of adult-onset Still’s disease: 1921-2021

et al. 2022

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ObjectivesThe aim of this research is to discuss the research status, hotspots, frontiers and development trends in the field of adult-onset Still’s disease (AOSD) based on bibliometrics and visual analysis by CiteSpace software.MethodsThe relevant research articles on AOSD from 1921 to 2021 were retrieved from the Scopus database. CiteSpace software was used to form a visual knowledge map and conduct analysis for the countries/regions, journals, authors, keywords, clusters, research hotspots and frontiers of the included articles.ResultsThere were 2,373 articles included, and the number of articles published during 1921-2021 is increasing. The country with the highest number of articles published was Japan (355, 14.96%), followed by the United States (329, 13.86%) and France (215, 9.06%). The author with the highest number of publications is Ansell, Barbara M. (30, 1.26%), and the author with the highest co-citation frequency is Yamaguchi, Masaya (703). Clinical Rheumatology is the journal with the highest publication frequency. The top five cluster groups were “joint”, “differential diagnosis”, “prednisolone”, “methotrexate” and “macrophage activation syndrome”. The diagnosis, treatment and pathogenesis of AOSD form the main research fields, and prognosis and complications are the research hotspots and trends.ConclusionsThe global research field in AOSD has expanded in the past 100 years. The complications and new pathogenesis of AOSD are hotspots in this field and need further study in the future.

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Treatment of refractory adult onset Still’s disease with tocilizumab—a single centre experience and literature review

Cited by 17 publications

References 41 publications

Biological treatment in resistant adult-onset Still’s disease: A single-center, retrospective cohort study

Biological treatment in resistant adult-onset Still’s disease: A single-center, retrospective cohort study

New insights on multigenic autoinflammatory diseases

Bibliometrics analysis on the research status and trends of adult-onset Still’s disease: 1921-2021

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