Abstract
BackgroundAdult-onset Still’s disease (AOSD) is a rare inflammatory disorder characterized by the classical triad of daily spiking fever, arthritis, and typical salmon-colored rash. Resistance to first-line corticosteroids and second-line disease modified anti-rheumatic-drugs defines refractory AOSD, which mostly includes the polycyclic or chronic courses of the disease. Anti-cytokine therapies are recommended in AOSD patients who are refractory to traditional treatments. This is the first report on the efficacy of colchicine in a patient with AOSD which was refractory to immunosuppressive treatments including biologics.Case presentationA 24-years Japanese female patient was referred to our hospital for the flare-up of AOSD under the combined treatments with steroid, immunosuppressants, and biologics. She was diagnosed with AOSD according to the Yamaguchi criteria, based on the presence of spiking fever, polyarthralgia, skin rash, and hyperferritinemia. Interleukin-6 or tumor necrosis factor-α blockade treatments were not effective, the oral administration of colchicine was stared under the immunosuppressive treatments with steroid and cyclosporine A (CyA). Colchicine treatment silenced the disease activity of AOSD. The dose of prednisolone was successfully tapered, and the elevated levels of C-reactive protein were normalized. Remission has been maintained for 13 months with the start of oral administration of colchicine.ConclusionWe concluded that colchicine is an alternative treatment in patients with refractory AOSD, particularly in those with impaired therapeutic effects against anti-cytokines therapies.
Highlights
Adult-onset Still’s disease (AOSD) is a rare inflammatory disorder characterized by the classical triad of daily spiking fever, arthritis, and typical salmon-colored rash
Discussions and conclusions Our patient is the first to be reported for effectiveness of colchicine in AOSD
The data available to date remain limited because of the rarity of the disease, it seems clear that use of colchicine represents a good alternative to biologics therapies, which can potentially cause adverse events including infections
Summary
Adult-onset Still’s disease (AOSD) is a rare inflammatory disorder characterized by the classical triad of daily spiking fever, arthritis, and typical salmon-colored rash. Conclusion: We concluded that colchicine is an alternative treatment in patients with refractory AOSD, in those with impaired therapeutic effects against anti-cytokines therapies. This is the first report on the successful induction of remission with colchicine in biologics-refractory AOSD. ESR erythrocyte sedimentation rate, sIL-2R soluble interleukin-2 receptor, Ig immunoglobulin, ANA anti-nuclearantibody, Anti-ds-DNA Ab anti-double stranded deoxyribonucleic acid antibody, SAA serum amyloid A, MMP-3 matrix metalloproteinase-3, HBs Ag hepatitis B virus surface antigen, HCV Ab anti-hepatitis C virus antibody, ASLO anti-streptolysin O, CMV cytomegalovirus prednisolone and methotrexate (16 mg/week).
Sign-in/Register to view highlights & summary Sign-in/Register to access full text options 
Talk to us
Join us for a 30 min session where you can share your feedback and ask us any queries you have
Schedule a callDisclaimer: All third-party content on this website/platform is and will remain the property of their respective owners and is provided on "as is" basis without any warranties, express or implied. Use of third-party content does not indicate any affiliation, sponsorship with or endorsement by them. Any references to third-party content is to identify the corresponding services and shall be considered fair use under The CopyrightLaw.
