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RESEARCH PRODUCT
Role of Colchicine Treatment in Tumor Necrosis Factor Receptor Associated Periodic Syndrome (TRAPS): Real-Life Data from the AIDA Network
Bruno FredianiLaura ObiciAlessandra RenieriJurgen SotaValeria CaggianoFrancesco LicciardiJosé Hernández-rodríguezRoberto GiacomelliPiero RuscittiAntonio VitaleNicola RiccoLorenzo DagnaCarlo SalvaraniMaria Cristina MaggioDavide MontinGiacomo EmmiAlessandra SorianoMarco CattaliniLuca CantariniOmbretta ViapianaVittoria LamacchiaFrancesco CasoRaffaele MannaAntonella Insalacosubject
Male0301 basic medicineEye DiseasesTRAPSColchicineAIDA NetworkGene mutationGastroenterologyReceptors Tumor Necrosis Factorchemistry.chemical_compoundSettore MED/38 - Pediatria Generale E Specialistica0302 clinical medicineReceptorsPathologyRB1-214ColchicineAge of OnsetYoung adultChildAmyloidosisAmyloidosisSyndromeMiddle AgedColchicine tumor necrosis factor TRAPSInflamacióPenetrancePhenotypeChild PreschoolFemaleJoint DiseasesResearch ArticleAdultRiskmedicine.medical_specialtyAdolescentFeverArticle SubjectImmunologyAdolescent; Adult; Age of Onset; Amyloidosis; Child; Child Preschool; Colchicine; Exanthema; Eye Diseases; Female; Fever; Humans; Joint Diseases; Male; Middle Aged; Mutation; Myalgia; Phenotype; Receptors Tumor Necrosis Factor; Retrospective Studies; Risk; Syndrome; Young AdultLower riskYoung Adult03 medical and health sciencesInternal medicinemedicineHumansPreschoolRetrospective StudiesInflammation030203 arthritis & rheumatologybusiness.industryTRAPSRetrospective cohort studyMyalgiaCell BiologyExanthemamedicine.disease030104 developmental biologychemistryMutationAge of onsetColchicineTumor Necrosis Factorbusinessdescription
Objective. To analyze the potential role of colchicine monotherapy in patients with tumor necrosis factor receptor associated periodic syndrome (TRAPS) in terms of control of clinical and laboratory manifestations. Methods. Patients with TRAPS treated with colchicine monotherapy were retrospectively enrolled; demographic, clinical and therapeutic data were collected and statistically analysed after having clustered patients according to different times at disease onset, penetrance of mutations, dosage of colchicine, and different disease manifestations. Results. 24 patients (14 males; 15 with pediatric disease onset) treated with colchicine monotherapy were enrolled. Colchicine resulted in a complete response in 3 (12.5%) cases, partial response in 14 (58.3%) patients, and lack of response in 7 (29.2%) patients. There were not significant differences in colchicine response between pediatric and adult disease onset (p=0.42), between low- and high-penetrance mutations (p=0.62), and according to different dosages (p=0.66). No significant differences were identified in the frequency of specific disease manifestations between patients experiencing any response to colchicine and patients with lack of response. Conclusions. Colchicine monotherapy is useful in a low percentage of TRAPS patients; nevertheless, it could be attempted in patients with milder phenotypes and at a lower risk of developing reactive amyloidosis.
| year | journal | country | edition | language |
|---|---|---|---|---|
| 2020-05-27 | Mediators of Inflammation |