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Longterm Beneficial Effect of Canakinumab in Colchicine-resistant Familial Mediterranean Fever.

TitleLongterm Beneficial Effect of Canakinumab in Colchicine-resistant Familial Mediterranean Fever.
Publication TypeJournal Article
Year of Publication2017
AuthorsLaskari, K., Boura P., Dalekos G. N., Garyfallos A., Karokis D., Pikazis D., Settas L., Skarantavos G., Tsitsami E., & Sfikakis P. P.
JournalJ Rheumatol
Volume44
Issue1
Pagination102-109
Date Published2017 01
ISSN0315-162X
KeywordsAdolescent, Adult, Aged, Anti-Inflammatory Agents, Antibodies, Monoclonal, Colchicine, Familial Mediterranean Fever, Female, Humans, Interleukin 1 Receptor Antagonist Protein, Male, Middle Aged, Off-Label Use, Remission Induction, Retreatment, Retrospective Studies, Treatment Failure, Treatment Outcome, Young Adult
Abstract

OBJECTIVE: To assess the efficacy and safety of the interleukin-1β (IL-1β) inhibitor canakinumab in all adolescent and adult patients with familial Mediterranean fever (FMF) identified from the Greek National Registry for off-label drug use between 2010 and 2015.METHODS: In this retrospective longitudinal outcome study, clinical and laboratory data were collected from 14 patients (7 men) aged median 38.5 years (range 13-70), with median disease duration of 14 years, and active FMF despite colchicine (n = 9) or both colchicine and anakinra (n = 5).RESULTS: All patients continued to receive canakinumab at last visit (median of 18 mos, range 13-53), which was initially given as monotherapy (n = 8) or in combination with colchicine and/or corticosteroids, every 4 (n = 7), 6 (n = 2), or 8 weeks (n = 5). Eleven patients (79%), including 6 receiving monotherapy, achieved complete clinical remission within 2 months (median), while normalization of all laboratory variables denoting inflammation occurred in 92% at 3 months (median). The remaining 3 patients achieved partial responses. Responses were sustained in all but 4 patients, who relapsed. Reducing the canakinumab administration interval from 8 or 6 weeks to 4 weeks led to suppression of disease activity in the relapsing patients. On the other hand, drug administration interval could be safely increased in 2 patients in remission. Corticosteroid doses were significantly reduced during followup. Canakinumab was well tolerated; 1 patient experienced a urinary tract infection and another one a viral gastroenteritis.CONCLUSION: Treatment with canakinumab in an individualized dosing scheme results in rapid and sustained remission in colchicine-resistant FMF.

DOI10.3899/jrheum.160518
Alternate JournalJ. Rheumatol.
PubMed ID28042127

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