Autoinflammatory & rare systemic diseases
Three-year follow up of canakinumab dose extension in children with colchicine resistant fasmilial Mediterranean fever: PeRA-RG Experience
Authors' conclusion
The dose extension protocol shows promising long-term outcomes in colchicine-resistant FMF.
Study context
- Population
- Children with colchicine-resistant FMF treated per a canakinumab dose extension protocol, Turkeymedian follow-up 47 months
- Participants
- 45 children
- Outcome
- Long-term outcomes of the dose extension protocol
Methodological detailsmulticentre, single-group, standardized dose extension protocol (Delphi consensus)
- Multicentre
- Single-group
- Standardized dose extension protocol (Delphi consensus)
Main finding
As reported in the abstract
- Attacks during bimonthly dosing, reverted to monthly dosing
- 7 patients (15.6%)
- Interval further extended to every 3 months
- 38 patients (84.4%)
- Returned from 3-monthly to bimonthly dosing due to attacks
- 11
- Treatment discontinued after remission on 3-month intervals; attack-free / had attacks
- 10; 5 / 5
- Continued 3-monthly dosing; lost to follow-up
- 17; 1
- Remained attack-free on 3-monthly dosing at median follow-up of 33 months (IQR 6.5)
- 16 (35.6%)
Observational data: associations, not effects.
Further findings
Clinical and laboratory findings, patients with vs without attacks
- Splenomegaly absent in the attack-free group
- P = 0.006
- Similar between patients with and without attacks
Original publication
Three-year follow up of canakinumab dose extension in children with colchicine resistant fasmilial Mediterranean fever: PeRA-RG Experience. Rheumatology (Oxford). Published 1 August 2025. doi:10.
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