ArticleRheumatology (Oxford, England)2025
Three-year follow-up of canakinumab dose extension in children with colchicine-resistant familial Mediterranean fever: PeRA-RG Experience.
Article in Rheumatology (Oxford, England), 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.
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1 citing paper in PubMed.
- Canakinumab dosing intervals in pediatric colchicine-resistant FMF: outcomes and predictors of successful extension in a real-world cohort.Rheumatology (Oxford, England) · 2026Article
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15 authors.
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Abstract
objectivesAnti-interleukin-1 therapies are effective for colchicine-resistant FMF, yet data on optimal duration in pediatric patients remain limited. A previous multicentre study showed favourable outcomes with a standardized canakinumab dose extension protocol, though follow-up was short. This study aimed to assess the long-term outcomes of this protocol.
methodsThe protocol, developed via a multicentre Delphi consensus in Turkey, recommends doubling the canakinumab dosing interval after 6 attack-free months and tripling it after 1 year of continued remission. This retrospective study included colchicine-resistant FMF patients treated according to the protocol, with data extracted from medical records.
resultsForty-five patients initiated monthly canakinumab. The median follow-up after starting canakinumab was 47 months (range 35-78). After 6 months, intervals were extended to every 2 months. During bimonthly dosing, 7 patients (15.6%) experienced attacks and reverted to monthly dosing. Of the 38 patients (84.4%) whose interval was further extended to every 3 months, 11 returned to bimonthly dosing due to attacks. Among 10 patients who achieved remission with 3-month intervals, treatment was discontinued; 5 remained attack-free, while 5 had attacks. Seventeen patients continued 3-monthly dosing, 1 was lost to follow-up and 16 (35.6%) remained attack-free at a median follow-up of 33 months (interquartile range: 6.5). Clinical and laboratory findings were similar between patients with and without attacks, except splenomegaly, which was absent in the attack-free group (p = 0.006).
conclusionThe dose extension protocol shows promising long-term outcomes in colchicine-resistant FMF. Larger, prospective studies are warranted to optimize treatment strategies.
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