5 years ago

A Nationwide Experience With The Off-label Use of Interleukin-1 Targeting Treatment in Familial Mediterranean Fever Patients

Fatos Onen, Ahmet Omma, Ismail Sari, Kenan Aksu, Sule Yavuz, Fatih Yildiz, Abdulsamet Erten, Gozde Yildirim, Omer Karadag, Atalay Dogru, Mehmet Engin Tezcan, Mehmet Ali Balci, Ahmet Mesut Onat, Yavuz Pehlivan, Orhan Kucuksahin, Bunyamin Kisacik, Metin Ozgen, Umut Kalyoncu, Timucin Kasifoglu, Omer Nuri Pamuk, Sibel Yilmaz Oner, Sukran Erten, Muhammed Cınar, Ayse Balkarli, Sedat Yilmaz, Fatma Alibaz Oner, Ali Ugur Unal, Pınar Cetin, Mehmet Sayarlıoglu, Gokhan Keser, Ferhat Oksuz, Haner Direskeneli, Abdurrahman Tufan, Servet Akar, Ozun Bayindir, Soner Senel
Objective Around 30–45% of patients with familial Mediterranean fever (FMF) have been reported to have attacks despite colchicine treatment. Currently, data on the treatment of colchicine-unresponsive or colchicine-intolerant FMF patients are limited; the most promising alternatives seem to be anti-interleukin-1 (anit-IL1) agents. Herein we report our experience with the off-label use of anti-IL1 agents in a large group of FMF patients. Methods In all, 21 centers from different geographical regions of Turkey were included in the current study. The medical records of all FMF patients who had used anti-IL1 treatment for at least 6 months were reviewed. Results In total, 172 FMF patients (83 [48%] female, mean age 36.2 [range, 18–68] years old) were included in the analysis; their mean age at symptom onset was 12.6 (range; 1–48) years, and the mean colchicine dose was 1.7 (0.5–4.0) mg/day. Of these patients, 151 were treated with anakinra and 21 with canakinumab. Anti-IL1 treatment was used because of colchicine-resistant disease in 84% and amyloidosis in 12% of subjects. During the mean 19.6 (6–98) months of treatment, the yearly attack frequency was significantly reduced (from 16.8 to 2.4; P < 0.001), and 42.1% of colchicine-resistant FMF patients were attack free. Serum levels of C-reactive protein, ESR, and 24 h urinary protein excretion (5458.7 mg/24 h before and 3557.3 mg/24 h after) were significantly reduced. Conclusion Anti-IL1 treatment is an effective alternative for controlling attacks and decreasing proteinuria in colchicine-resistant FMF patients. This article is protected by copyright. All rights reserved.

Publisher URL: http://onlinelibrary.wiley.com/resolve/doi

DOI: 10.1002/acr.23446

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