Successful tocilizumab therapy in seven patients with refractory adult-onset Still's disease

Bannai, Ei; Yamashita, Hiroyuki; Kaneko, Shunta; Ueda, Yoshimichi; Ozaki, Takashi; Tsuchiya, Haruka; Takahashi, Yuko; Kaneko, Hiroshi; Kano, Toshikazu; Matsubara, Akio

doi:10.3109/14397595.2014.899178

Cited by 34 publications

(22 citation statements)

References 22 publications

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“…Since aseptic abscess is a known complication of SAPHO syndrome, TCZ treatment in our patient might have induced the aseptic abscess via exacerbating SAPHO syndrome. Indeed, in adult‐onset Still's disease (AOSD) or systemic juvenile idiopathic arthritis (sJIA), TCZ has been reported to be associated with macrophage activation syndrome (MAS), which is a serious complication of AOSD or sJIA . As previously reported, a transient increase in the serum levels of IL‐6 immediately after IL‐6 receptor blockade might induce MAS in AOSD or sJIA or induce aseptic abscess in SAPHO syndrome.…”

Section: Cytokine Serum Levels In a Patient With Sapho Syndrome At Onmentioning

confidence: 92%

“…Indeed, in adult‐onset Still's disease (AOSD) or systemic juvenile idiopathic arthritis (sJIA), TCZ has been reported to be associated with macrophage activation syndrome (MAS), which is a serious complication of AOSD or sJIA . As previously reported, a transient increase in the serum levels of IL‐6 immediately after IL‐6 receptor blockade might induce MAS in AOSD or sJIA or induce aseptic abscess in SAPHO syndrome. We now recognize that our initial treatment method was wrong.…”

Section: Cytokine Serum Levels In a Patient With Sapho Syndrome At Onmentioning

confidence: 92%

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Development of aseptic subcutaneous abscess after tocilizumab therapy in a patient with SAPHO syndrome complicated by amyloid A amyloidosis

et al. 2014

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Section: Cytokine Serum Levels In a Patient With Sapho Syndrome At Onmentioning

confidence: 92%

Section: Cytokine Serum Levels In a Patient With Sapho Syndrome At Onmentioning

confidence: 92%

Development of aseptic subcutaneous abscess after tocilizumab therapy in a patient with SAPHO syndrome complicated by amyloid A amyloidosis

et al. 2014

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“…It is characterized by elevated levels of inflammatory cytokines in serum including IL-6, IL-18, IL-1, TNFα and IFNγ. An increasing number of case reports or series has shown the effectiveness of TCZ on AOSD and reduction of glucocorticoid dose [ 117 , 118 , 119 ]. A meta-analysis of 10 studies with 147 patients showed that TCZ therapy achieved a partial remission rate in 85.38% and complete remission rate in 77.91%, and even in patients with refractory cases, the remission rate was 87.92% [ 120 ], suggesting a promising effect of TCZ on AOSD.…”

Section: Therapeutic Targeting Of Il-6 In Diseasesmentioning

confidence: 99%

The Two-Faced Cytokine IL-6 in Host Defense and Diseases

Narazaki

Kishimoto

2018

IJMS

165

129

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Interleukein-6 (IL-6), is produced locally from infectious or injured lesions and is delivered to the whole body via the blood stream, promptly activating the host defense system to perform diverse functions. However, excessive or sustained production of IL-6 is involved in various diseases. In diseases, the IL-6 inhibitory strategy begins with the development of the anti-IL-6 receptor antibody, tocilizumab (TCZ). This antibody has shown remarkable effects on Castleman disease, rheumatoid arthritis and juvenile idiopathic arthritis. In 2017, TCZ was proven to work effectively against giant cell arteritis, Takayasu arteritis and cytokine releasing syndrome, initiating a new era for the treatment of these diseases. In this study, the defensive functions of IL-6 and various pathological conditions are compared. Further, the diseases of which TCZ has been approved for treatment are summarized, the updated results of increasing off-label use of TCZ for various diseases are reviewed and the conditions for which IL-6 inhibition might have a beneficial role are discussed. Given the involvement of IL-6 in many pathologies, the diseases that can be improved by IL-6 inhibition will expand. However, the important role of IL-6 in host defense should always be kept in mind in clinical practice.

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“…Increased levels of interleukin (IL)‐6 and other pro‐inflammatory cytokines have been shown in AOSD. Therefore, tocilizumab (TCZ), an IL‐6 receptor antagonist monoclonal antibody, has been tried in AOSD patients and successful results were reported 6‐17 . Most of the studies evaluating the treatment of TCZ are case reports involving a small number of patients, and there are several single‐centre or multi‐centre studies 6‐17 .…”

Section: Introductionmentioning

confidence: 99%

Adult‐onset still’s disease and treatment results with tocilizumab

2020

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Aims Adult‐onset Still's disease (AOSD) is a rare and non‐familial auto‐inflammatory disorder. Increased levels of IL‐6 and other pro‐inflammatory cytokines have been shown in AOSD. To evaluate the efficacy and safety profile of tocilizumab (TCZ), an IL‐6 receptor antagonist monoclonal antibody, in AOSD. Methods Thirty‐nine patients followed up with the diagnosis of AOSD between 2013 and 2019 were retrospectively evaluated and the 16 patients (10 Female/6 Male) treated with TCZ for refractory AOSD were included in the study group. Among the remaining 23 patients 16 had non‐biological treatments and had no important complications at the presentation. TCZ was given to patients at a dose of 4‐8 mg/kg every 4 weeks. Patients were evaluated after 3‐6 months of TCZ treatment for side effects, inflammatory and clinical response and concomitant treatments. Results In TCZ (+) patients, the majority were female (62.5%), the mean age at disease onset was 38.5 ± 17.9 (20‐81) years, and the most common symptoms and signs were myalgia (81.3%), fever (81.3%) and skin eruptions (75%). There was no difference between TCZ (+) and TCZ (−) groups for age, sex and clinical presentations. There was a significant decrease in dose of prednisolone, sedimentation rate, leucocyte count, C‐reactive protein and ferritin levels and improvement in all clinical complaints after TCZ treatment. There were no relapses during the treatment. Three patients are in remission and under follow‐up without any treatment after cessation of TCZ (4 months‐3 years). No exacerbation of disease yet seen in those patients. Conclusions TCZ is an effective and well‐tolerated treatment option for treatment resistant AOSD and contributes to the glucocorticoid‐sparing. Since TCZ is a new drug in the treatment of AOSD, further studies are needed to assess whether the complications reported during the treatment are because of TCZ or natural course of the disease or coincidental findings.

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Successful tocilizumab therapy in seven patients with refractory adult-onset Still's disease

Cited by 34 publications

References 22 publications

Development of aseptic subcutaneous abscess after tocilizumab therapy in a patient with SAPHO syndrome complicated by amyloid A amyloidosis

Development of aseptic subcutaneous abscess after tocilizumab therapy in a patient with SAPHO syndrome complicated by amyloid A amyloidosis

The Two-Faced Cytokine IL-6 in Host Defense and Diseases

Adult‐onset still’s disease and treatment results with tocilizumab

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