Servet Akar1, Pınar Cetin2, Umut Kalyoncu3, Omer Karadag3, Ismail Sari4, Muhammed Cınar5, Sedat Yilmaz5, Ahmet Mesut Onat6, Bunyamin Kisacik6, Abdulsamet Erden3, Ayse Balkarli7, Orhan Kucuksahin8, Sibel Yilmaz Oner9, Soner Senel10, Abdurrahman Tufan11, Haner Direskeneli12, Ferhat Oksuz13, Yavuz Pehlivan14, Ozun Bayindir15, Gokhan Keser15, Kenan Aksu15, Ahmet Omma16, Timucin Kasifoglu17, Ali Ugur Unal12, Fatih Yildiz18, Mehmet Ali Balci19, Sule Yavuz20, Sukran Erten8, Metin Ozgen21, Mehmet Sayarlıoglu21, Atalay Dogru22, Gozde Yildirim23, Fatma Alibaz Oner24, Mehmet Engin Tezcan25, Omer Nuri Pamuk19, Fatos Onen4. 1. Katip Celebi University Hospital, Izmir, Turkey. 2. Dumlupinar University Evliya Celebi Hospital, Kutahya, Turkey. 3. Hacettepe University Hospital, Ankara, Turkey. 4. Dokuz Eylul University Hospital, Izmir, Turkey. 5. Gulhane Military Hospital, Ankara, Turkey. 6. Gaziantep University Hospital, Gaziantep, Turkey. 7. Antalya Training and Research Hospital, Antalya, Turkey. 8. Yildirim Beyazit University Hospital, Ankara, Turkey. 9. Bakirkoy Dr. Sadi Konuk Training and Research Hospital, Istanbul, Turkey. 10. Erciyes University Hospital, Kayseri, Turkey. 11. Gazi University Hospital, Ankara, Turkey. 12. Marmara University Hospital, Istanbul, Turkey. 13. Mersin Training and Research Hospital, Mersin, Turkey. 14. Uludag University Hospital, Bursa, Turkey. 15. Ege University Hospital, İzmir, Turkey. 16. Numune Training and Research Hospital, Ankara, Turkey. 17. Eskisehir Osmangazi University Hospital, Eskisehir, Turkey. 18. Van Training and Research Hospital, Van, Turkey. 19. Trakya University Hospital, Edirne, Turkey. 20. Bilim University Hospital, Istanbul, Turkey. 21. Ondokuz Mayis University Hospital, Samsun, Turkey. 22. Suleyman Demirel University Hospital, Isparta, Turkey. 23. Sutcu Imam University Hospital, Kahramanmaras, Turkey. 24. Fatih Sultan Mehmet Training and Research Hospital, Istanbul, Turkey. 25. Kartal Dr. Lutfi Kirdar Training and Research Hospital, Istanbul, Turkey.
Abstract
OBJECTIVE: Approximately 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 (anti-IL-1) agents. Here we report our experience with the off-label use of anti-IL-1 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-IL-1 treatment for at least 6 months were reviewed. RESULTS: In total, 172 FMF patients (83 [48%] female, mean age 36.2 years [range 18-68]) were included in the analysis; mean age at symptom onset was 12.6 years (range 1-48), and the mean colchicine dose was 1.7 mg/day (range 0.5-4.0). Of these patients, 151 were treated with anakinra and 21 with canakinumab. Anti-IL-1 treatment was used because of colchicine-resistant disease in 84% and amyloidosis in 12% of subjects. During the mean 19.6 months of treatment (range 6-98), 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, erythrocyte sedimentation rate, and 24-hour urinary protein excretion (5,458.7 mg/24 hours before and 3,557.3 mg/24 hours after) were significantly reduced. CONCLUSION: Anti-IL-1 treatment is an effective alternative for controlling attacks and decreasing proteinuria in colchicine-resistant FMF patients.
OBJECTIVE: Approximately 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 FMFpatients are limited; the most promising alternatives seem to be anti-interleukin-1 (anti-IL-1) agents. Here we report our experience with the off-label use of anti-IL-1 agents in a large group of FMFpatients. METHODS: In all, 21 centers from different geographical regions of Turkey were included in the current study. The medical records of all FMFpatients who had used anti-IL-1 treatment for at least 6 months were reviewed. RESULTS: In total, 172 FMFpatients (83 [48%] female, mean age 36.2 years [range 18-68]) were included in the analysis; mean age at symptom onset was 12.6 years (range 1-48), and the mean colchicine dose was 1.7 mg/day (range 0.5-4.0). Of these patients, 151 were treated with anakinra and 21 with canakinumab. Anti-IL-1 treatment was used because of colchicine-resistant disease in 84% and amyloidosis in 12% of subjects. During the mean 19.6 months of treatment (range 6-98), the yearly attack frequency was significantly reduced (from 16.8 to 2.4; P < 0.001), and 42.1% of colchicine-resistant FMFpatients were attack free. Serum levels of C-reactive protein, erythrocyte sedimentation rate, and 24-hour urinary protein excretion (5,458.7 mg/24 hours before and 3,557.3 mg/24 hours after) were significantly reduced. CONCLUSION: Anti-IL-1 treatment is an effective alternative for controlling attacks and decreasing proteinuria in colchicine-resistant FMFpatients.
Authors: T Kallinich; N Blank; T Braun; E Feist; U Kiltz; U Neudorf; P T Oommen; C Weseloh; H Wittkowski; J Braun Journal: Z Rheumatol Date: 2019-02 Impact factor: 1.372
Authors: Michael Boehm; Eva Nora Bukosza; Nicole Huttary; Rebecca Herzog; Christoph Aufricht; Klaus Kratochwill; Christoph A Gebeshuber Journal: PLoS One Date: 2019-03-28 Impact factor: 3.240