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Efficacy and long-term follow-up of IL-1R inhibitor anakinra in adults with Stills disease a case-series study.doc

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Efficacy and long-term follow-up of IL-1R inhibitor anakinra in adults with Stills disease a case-series study

Laskarietal.ArthritisResearchTherapy2011,13:R91 /content/13/3/R91 RESEARCH ARTICLE OpenAccess Efficacyandlong-termfollow-upofIL-1R inhibitoranakinrainadultswithStill’ sdisease: acase-seriesstudy KaterinaLaskari*,AthanasiosGTzioufasandHaralamposMMoutsopoulos Abstract Introduction:Toassesstheefficacyandsafetyoftheinterleukin-1receptor(IL-1R)inhibitoranakinrainadult patientswithrefractoryStill’ sdisease. Methods:Twenty-fivepatients(13malesand12females,medianage32years,mediandiseasedurationseven months)withStill’sdiseaseweretreatedwithsubcutaneousinjectionsofanakinra(100mg/day).Treatmentwas givenasadjuncttherapyin16patientsandasstandalonein9patientsforamediantimeof15months(range 1.5-71).Theclinicalandlaboratoryparametersduringfollow-upwererecorded. Results:In84%ofpatientstheclinicalactivityresolvedcompletelywithinafewdays(mediantime0.2months), andresponsewasmaintaineduntilthelastvisitinallbutonepatient.Acompleteresponseofalldisease-related symptoms(clinicalandlaboratory)occurredsubsequentlywithinamediantimeofthreemonthsin80%of patients.Apartialclinicalandlaboratoryimprovementwasshownin12%and16%ofpatients,respectively.The VisualizedAnalogueScaleandHealthAssessmentQuestionnairescoressignificantlydecreasedduringtreatment. TheproportionofpatientsachievingtheAmericanCollegeofRheumatology20(ACR20)score(20%improvement) was82%atonemonthandimprovedto100%atoneyear.Themeanoralcorticosteroiddosewassignificantly reducedateachvisit.Anakinrawasdiscontinuedduetounresponsivenessinonepatientandduetorelapsing diseaseinanother.Treatmentwasalsowithdrawninthreepatientswithsevereskinreactions(urticaria).Seven patientsexperiencedaninfectionduringfollow-up. Conclusions:Therapidandsustainedresponseinthemajorityofourpatientsencouragestheuseofanakinrain adultswithStill’ sdisease. Introduction Immunosuppressants,mainlymethotrexate(MTX),used Still’sdiseaseisararesystemicinflammatorydisorderof as steroid-sparing drugs, have shown modest efficacy unknown etiology characterized by quotidian spi