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

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Abstract

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.

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Acknowledgements

Language editing was done by Scribendi Language editing services.

Funding

This paper was not funded by anyone.

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Authors and Affiliations

  1. Division of Rheumatology, Department of Internal Medicine, Gazi University Faculty of Medicine, 06100, Ankara, Turkey

    Hazan Karadeniz, Aslıhan Avanoğlu Güler, Nuh Atas, Hasan Satış, Reyhan Bilici Salman, Hakan Babaoglu & Abdurrahman Tufan

Authors
  1. Hazan Karadeniz
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  2. Aslıhan Avanoğlu Güler
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  3. Nuh Atas
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  4. Hasan Satış
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  5. Reyhan Bilici Salman
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  6. Hakan Babaoglu
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  7. Abdurrahman Tufan
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Contributions

HK and AT designed the study and wrote the initial draft. NA, HB, RBS, AAG and HS contributed to the interpretation of data and revised it critically. All authors contributed to data collection. All authors approved the version to be published and agreed to be accountable for all aspects of the work.

Corresponding author

Correspondence to Hazan Karadeniz.

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Conflict of ınterest

AT is Consultant for: Novartis and Abdi Ibrahim, Speaker Bureau of: Novartis, Pfizer and UCB with no conflict related to this study. AT is editorial board member of two international journals and reviewer for 3 international journals and research funding bodies. None of the authors have conflict of interest and non-financial relationships with agencies promoting tofacitinib.

Informed consent

Written informed consent was obtained from all subjects. Permissions was taken from regulatory agencies for off-label uses of drugs. For each patient, we applied for permission of use and shared our Patient files with Medical Devices Agency of the Turkish Ministry of Health. The Turkish Ministry of Health granted the compassionate use of tofacitinib for FMF patients with special permission.

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Karadeniz, H., Güler, A.A., Atas, N. et al. Tofacitinib for the treatment for colchicine-resistant familial Mediterranean fever: case-based review. Rheumatol Int 40, 169–173 (2020). https://doi.org/10.1007/s00296-019-04490-7

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  • DOI: https://doi.org/10.1007/s00296-019-04490-7

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