Session Information
Session Type: Abstract Submissions (ACR)
Background/Purpose
Adult onset Still’s disease (AOSD) is a rare inflammatory disorder of unknown etiology. Refractory cases to conventional therapy require biologic agents. Although IL-1 targeting therapy is very effective, but tocilizumab (TCZ), an anti-interleukin-6 receptor monoclonal antibody may be an effective agent especially in the countries where anti-IL-1 therapy is not easily available. In this study, we assessed the efficacy of TCZ in multicenter retrospective fashion.
Methods
We retrospectively collected clinical data of AOSD patients who were treated with in Korea. The response to TCZ was defined as decreased modified Pouchot’s score more than 2 score with decreased acute phase reactants compared to initial treatment of TCZ at least two consecutive months.
Results
Patients (15 women/5 men) had a mean age of 42.5± 18.8 years and the age of diagnosis was 37.3 ±21.2years old. The mean disease duration before TCZ treatment was 40.7 ± 33.6 months. Ten patients (52.6%) had polycyclic systemic pattern and 9 patient had chronic articular pattern, but 1 patient was no classified due to short disease duration.
Immune modulating agents before TCZ therapy were as follows: methotrexate (n=18), leflunomide (n=12), cyclosporine (n=9), azathioprine (n=8) and hydroxychloroquine (n=7). Biologic agents before TCZ therapy were etanercept (n=7), infliximab (n=4), adalimumab (n=3), abatacept (n=2) and anakinra (n=1). At TCZ onset, the most frequent clinical manifestations were: joint manifestations (80.0%), rash (50.0%), myalgia (35.0%), fever (25.0%) and sore throat (25.0%) (Table 1). The baseline laboratory parameters such as leukocytosis, ESR, CRP and serum ferritin showed high level (Table 1). The mean dose of TCZ was 7.8 mg/kg (4-8 mg/kg) per 4 weeks and mean duration of TCZ administration was 7.9 ± 6.7 months. The 90.0% of patients showed clinical and laboratory improvement after TCZ therapy (Table 1). The median dose of prednisolone was also significantly reduced from17.1± 10.1 mg/day at TCZ onset to 7.5 ± 3.9 mg/day at 12months (Table 1).
The 35% of patients had side effects during TCZ treatment as follows: leukopenia (n=2), hypertension (n=2), alopecia (n=2), pneumonia (n=1). Four patients (20%) relapsed after 5.0 ± 3.6 months of discontinuation of TCZ.
Conclusion
TCZ was effective in Korean AOSD patients refractory to conventional therapy and/or other biologic agents. Larger and prospective study is needed for further investigation.
Table 1. Change of clinical manifestations and laboratory parameters before and after TCZ treatment
|
|
Baseline (N=20) |
After 6 months (N=12) |
After 12 months (N=7) |
|
Fever (n, %) |
5, 25.0 |
0, 0 |
1, 14.3 |
|
Sore throat (n, %) |
5, 25.0 |
1, 8.3 |
0, 0 |
|
Rash* (n, %) |
10, 50.0 |
1, 8.3 |
0, 0 |
|
Itching (n, %) |
5, 25.0 |
1, 8.3 |
0, 0 |
|
Myalgia* (n, %) |
7, 35.0 |
0, 8.3 |
0, 0 |
|
Arthralgia/arthritis* (n, %) |
16, 80.0 |
5, 66.7 |
1, 14.3 |
|
Lymphadenopathy (n, %) |
1, 5.0 |
0, 0 |
0, 0 |
|
Splenomegaly (n, %) |
0, 0 |
0, 0 |
0, 0 |
|
Serositis (n, %) |
1, 5.0 |
0, 0 |
0, 0 |
|
Modified Pouchot’s Score, mean ± SD |
3.1 ± 1.7 |
1.3 ± 1.3 |
1.4 ± 1.0 |
|
Leukocytosis, mean ± SD |
12697.5 ± 5193.4 |
11130.8 ± 6221.7 |
11547.1 ± 6935.3 |
|
ESR (mm/hour), mean ± SD** |
63.6 ± 28.0 |
10.3± 15.8 |
14.0 ± 23.3 |
|
CRP (mg/dL), mean ± SD* |
4.4 ± 3.6 |
1.0± 2.2 |
2.3 ± 2.9 |
|
Ferritin (ng/mL), mean ± SD |
1657.8 ± 2222.6 |
558.4± 1113.4 |
165.2 ± 130.4 |
|
Prednisolone dose (mg/day), mean ± SD* |
17.1 ± 10.1 |
7.1 ± 5.1 |
7.5 ± 3.9 |
*P < 0.05, ** P < 0.001
Disclosure:
J. J. Kim,
None;
J. H. Lee,
None;
C. N. Son,
None;
H. A. Kim,
None;
K. H. Lee,
None;
S. T. Choi,
None;
E. Y. Lee,
None;
K. C. Shin,
None;
H. S. Cha,
None;
D. H. Yoo,
None.
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