Role of Colchicine Treatment in Tumor Necrosis Factor Receptor Associated Periodic Syndrome (TRAPS): Real-Life Data from the AIDA Network

Author:

Vitale Antonio1ORCID,Sota Jurgen1ORCID,Obici Laura2,Ricco Nicola1,Maggio Maria Cristina3,Cattalini Marco4,Ruscitti Piero5,Caso Francesco6ORCID,Manna Raffaele7ORCID,Viapiana Ombretta8,Caggiano Valeria1,Emmi Giacomo9,Insalaco Antonella10,Montin Davide11,Licciardi Francesco11,Soriano Alessandra12,Dagna Lorenzo13,Salvarani Carlo12,Lamacchia Vittoria1415,Hernández-Rodríguez José16,Giacomelli Roberto5,Frediani Bruno1,Renieri Alessandra1415ORCID,Cantarini Luca1ORCID

Affiliation:

1. Research Center of Systemic Autoinflammatory Diseases and Behçet's Disease and Rheumatology-Ophthalmology Collaborative Uveitis Center, Department of Medical Sciences, Surgery and Neurosciences, University of Siena, Italy

2. Amyloidosis Research and Treatment Center, Fondazione IRCCS Policlinico San Matteo, Pavia, Italy

3. Department of Health Promotion Sciences Maternal and Infantile Care, Internal Medicine and Medical Specialties “G. D’Alessandro”, University of Palermo, Palermo, Italy

4. Pediatric Clinic, University of Brescia and Spedali Civili di Brescia, Brescia, Italy

5. Division of Rheumatology, Department of Biotechnological and Applied Clinical Science, University of L’Aquila, L’Aquila, Italy

6. Rheumatology Unit, Department of Clinical Medicine and Surgery, School of Medicine, University Federico II of Naples, Naples, Italy

7. Institute of Internal Medicine, Fondazione Policlinico Universitario A. Gemelli IRCCS, Rome, Italy

8. Rheumatology Section, Department of Medicine, University of Verona, Verona, Italy

9. Department of Experimental and Clinical Medicine, University of Florence, Florence, Italy

10. Division of Rheumatology, Department of Pediatric Medicine, Bambino Gesù Children’s Hospital IRCCS, Rome, Italy

11. Division of Pediatric Immunology and Rheumatology, Department of Public Health and Pediatrics, “Regina Margherita” Children Hospital, University of Turin, Turin, Italy

12. Azienda Unità Sanitaria Locale-IRCCS di Reggio Emilia, Reggio Emilia, Italy

13. Unit of Immunology, Rheumatology, Allergy and Rare Diseases, IRCCS San Raffaele Hospital, Milan, Italy

14. Medical Genetics, University of Siena, Siena, Italy

15. Genetica Medica, Azienda Ospedaliera Universitaria Senese, Siena, Italy

16. Vasculitis Research Unit and Autoinflammatory Diseases Clinical Unit, Department of Autoimmune Diseases, Hospital Clinic of Barcelona, IDIBAPS, University of Barcelona, Barcelona, Spain

Abstract

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.

Publisher

Hindawi Limited

Subject

Cell Biology,Immunology

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