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

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...

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Main Authors: Antonio Vitale, Jurgen Sota, Laura Obici, Nicola Ricco, Maria Cristina Maggio, Marco Cattalini, Piero Ruscitti, Francesco Caso, Raffaele Manna, Ombretta Viapiana, Valeria Caggiano, Giacomo Emmi, Antonella Insalaco, Davide Montin, Francesco Licciardi, Alessandra Soriano, Lorenzo Dagna, Carlo Salvarani, Vittoria Lamacchia, José Hernández-Rodríguez, Roberto Giacomelli, Bruno Frediani, Alessandra Renieri, Luca Cantarini
Format: Article
Language:English
Published: Wiley 2020-01-01
Series:Mediators of Inflammation
Online Access:http://dx.doi.org/10.1155/2020/1936960
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author Antonio Vitale
Jurgen Sota
Laura Obici
Nicola Ricco
Maria Cristina Maggio
Marco Cattalini
Piero Ruscitti
Francesco Caso
Raffaele Manna
Ombretta Viapiana
Valeria Caggiano
Giacomo Emmi
Antonella Insalaco
Davide Montin
Francesco Licciardi
Alessandra Soriano
Lorenzo Dagna
Carlo Salvarani
Vittoria Lamacchia
José Hernández-Rodríguez
Roberto Giacomelli
Bruno Frediani
Alessandra Renieri
Luca Cantarini
author_facet Antonio Vitale
Jurgen Sota
Laura Obici
Nicola Ricco
Maria Cristina Maggio
Marco Cattalini
Piero Ruscitti
Francesco Caso
Raffaele Manna
Ombretta Viapiana
Valeria Caggiano
Giacomo Emmi
Antonella Insalaco
Davide Montin
Francesco Licciardi
Alessandra Soriano
Lorenzo Dagna
Carlo Salvarani
Vittoria Lamacchia
José Hernández-Rodríguez
Roberto Giacomelli
Bruno Frediani
Alessandra Renieri
Luca Cantarini
author_sort Antonio Vitale
collection DOAJ
description 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.
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spelling doaj-art-9be110f572054fa49baef19be71ded152025-02-03T05:44:13ZengWileyMediators of Inflammation0962-93511466-18612020-01-01202010.1155/2020/19369601936960Role of Colchicine Treatment in Tumor Necrosis Factor Receptor Associated Periodic Syndrome (TRAPS): Real-Life Data from the AIDA NetworkAntonio Vitale0Jurgen Sota1Laura Obici2Nicola Ricco3Maria Cristina Maggio4Marco Cattalini5Piero Ruscitti6Francesco Caso7Raffaele Manna8Ombretta Viapiana9Valeria Caggiano10Giacomo Emmi11Antonella Insalaco12Davide Montin13Francesco Licciardi14Alessandra Soriano15Lorenzo Dagna16Carlo Salvarani17Vittoria Lamacchia18José Hernández-Rodríguez19Roberto Giacomelli20Bruno Frediani21Alessandra Renieri22Luca Cantarini23Research 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, ItalyResearch 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, ItalyAmyloidosis Research and Treatment Center, Fondazione IRCCS Policlinico San Matteo, Pavia, ItalyResearch 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, ItalyDepartment of Health Promotion Sciences Maternal and Infantile Care, Internal Medicine and Medical Specialties “G. D’Alessandro”, University of Palermo, Palermo, ItalyPediatric Clinic, University of Brescia and Spedali Civili di Brescia, Brescia, ItalyDivision of Rheumatology, Department of Biotechnological and Applied Clinical Science, University of L’Aquila, L’Aquila, ItalyRheumatology Unit, Department of Clinical Medicine and Surgery, School of Medicine, University Federico II of Naples, Naples, ItalyInstitute of Internal Medicine, Fondazione Policlinico Universitario A. Gemelli IRCCS, Rome, ItalyRheumatology Section, Department of Medicine, University of Verona, Verona, ItalyResearch 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, ItalyDepartment of Experimental and Clinical Medicine, University of Florence, Florence, ItalyDivision of Rheumatology, Department of Pediatric Medicine, Bambino Gesù Children’s Hospital IRCCS, Rome, ItalyDivision of Pediatric Immunology and Rheumatology, Department of Public Health and Pediatrics, “Regina Margherita” Children Hospital, University of Turin, Turin, ItalyDivision of Pediatric Immunology and Rheumatology, Department of Public Health and Pediatrics, “Regina Margherita” Children Hospital, University of Turin, Turin, ItalyAzienda Unità Sanitaria Locale-IRCCS di Reggio Emilia, Reggio Emilia, ItalyUnit of Immunology, Rheumatology, Allergy and Rare Diseases, IRCCS San Raffaele Hospital, Milan, ItalyAzienda Unità Sanitaria Locale-IRCCS di Reggio Emilia, Reggio Emilia, ItalyMedical Genetics, University of Siena, Siena, ItalyVasculitis Research Unit and Autoinflammatory Diseases Clinical Unit, Department of Autoimmune Diseases, Hospital Clinic of Barcelona, IDIBAPS, University of Barcelona, Barcelona, SpainDivision of Rheumatology, Department of Biotechnological and Applied Clinical Science, University of L’Aquila, L’Aquila, ItalyResearch 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, ItalyMedical Genetics, University of Siena, Siena, ItalyResearch 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, ItalyObjective. 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.http://dx.doi.org/10.1155/2020/1936960
spellingShingle Antonio Vitale
Jurgen Sota
Laura Obici
Nicola Ricco
Maria Cristina Maggio
Marco Cattalini
Piero Ruscitti
Francesco Caso
Raffaele Manna
Ombretta Viapiana
Valeria Caggiano
Giacomo Emmi
Antonella Insalaco
Davide Montin
Francesco Licciardi
Alessandra Soriano
Lorenzo Dagna
Carlo Salvarani
Vittoria Lamacchia
José Hernández-Rodríguez
Roberto Giacomelli
Bruno Frediani
Alessandra Renieri
Luca Cantarini
Role of Colchicine Treatment in Tumor Necrosis Factor Receptor Associated Periodic Syndrome (TRAPS): Real-Life Data from the AIDA Network
Mediators of Inflammation
title Role of Colchicine Treatment in Tumor Necrosis Factor Receptor Associated Periodic Syndrome (TRAPS): Real-Life Data from the AIDA Network
title_full Role of Colchicine Treatment in Tumor Necrosis Factor Receptor Associated Periodic Syndrome (TRAPS): Real-Life Data from the AIDA Network
title_fullStr Role of Colchicine Treatment in Tumor Necrosis Factor Receptor Associated Periodic Syndrome (TRAPS): Real-Life Data from the AIDA Network
title_full_unstemmed Role of Colchicine Treatment in Tumor Necrosis Factor Receptor Associated Periodic Syndrome (TRAPS): Real-Life Data from the AIDA Network
title_short Role of Colchicine Treatment in Tumor Necrosis Factor Receptor Associated Periodic Syndrome (TRAPS): Real-Life Data from the AIDA Network
title_sort role of colchicine treatment in tumor necrosis factor receptor associated periodic syndrome traps real life data from the aida network
url http://dx.doi.org/10.1155/2020/1936960
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