Tofacitinib for the treatment for colchicine-resistant familial Mediterranean fever: case-based review

Familial Mediterranean fever is characterized by self-limited attacks of serositis and arthritis. However, substantial number of patients suffer from chronic complications of this disease, primarily involving musculoskeletal system. Treatment for these complications is challenging due to limited evi...

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Bibliographic Details
Published in:Rheumatology international 2020, Vol.40 (1), p.169-173
Main Authors: Karadeniz, Hazan, Güler, Aslıhan Avanoğlu, Atas, Nuh, Satış, Hasan, Salman, Reyhan Bilici, Babaoglu, Hakan, Tufan, Abdurrahman
Format: Article
Language:English
Subjects:
Adult
Antibodies, Monoclonal, Humanized - therapeutic use
Antirheumatic Agents - therapeutic use
Arthritis
Case Based Review
Colchicine - therapeutic use
Drug Resistance
Familial Mediterranean Fever - drug therapy
Female
Humans
Interleukin 1 Receptor Antagonist Protein - therapeutic use
Janus Kinase Inhibitors - therapeutic use
Male
Medicine
Medicine & Public Health
Middle Aged
Piperidines - therapeutic use
Pyrimidines - therapeutic use
Rheumatology
Treatment Failure
Treatment Outcome
Tubulin Modulators - therapeutic use
Tumor Necrosis Factor Inhibitors - therapeutic use
Tumor necrosis factor-TNF
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Summary:Familial Mediterranean fever is characterized by self-limited attacks of serositis and arthritis. However, substantial number of patients suffer from chronic complications of this disease, primarily involving musculoskeletal system. Treatment for these complications is challenging due to limited evidence. Interleukin-1 (IL-1) antagonists, tocilizumab and anti-tumor necrosis factor (anti-TNF) agents are off-label treatment options for the management of chronic manifestations of FMF, such as secondary (AA) amyloidosis, chronic arthritis and sacroiliitis. This paper presents a case series of four FMF patients who are refractory to IL-1 antagonists, anti-TNF agents and tocilizumab, who responded well to tofacitinib. The authors also conducted a comprehensive literature search for studies investigating tofacitinib use in FMF patients. Although still limited, current data suggest that tofacitinib could be a useful treatment option for FMF patients with associated inflammatory comorbid conditions and chronic manifestations of disease.
ISSN:0172-8172
1437-160X
DOI:10.1007/s00296-019-04490-7