Biotechnological Agents for Patients With Tumor Necrosis Factor Receptor Associated Periodic Syndrome—Therapeutic Outcome and Predictors of Response: Real-Life Data From the AIDA Network
Objective: To describe the role of biotechnological therapies in patients with tumor necrosis factor receptor associated periodic syndrome (TRAPS) and to identify any predictor of complete response.Methods: Clinical, laboratory, and therapeutic data from 44 Caucasian TRAPS patients treated with biol...
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Frontiers Media S.A.
2021-07-01
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| Series: | Frontiers in Medicine |
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| Online Access: | https://www.frontiersin.org/articles/10.3389/fmed.2021.668173/full |
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| author | Antonio Vitale Laura Obici Marco Cattalini Giuseppe Lopalco Giampaolo Merlini Nicola Ricco Alessandra Soriano Francesco La Torre Elena Verrecchia Antonella Insalaco Lorenzo Dagna Masen Abdel Jaber Davide Montin Giacomo Emmi Luisa Ciarcia Sara Barneschi Paola Parronchi Piero Ruscitti Maria Cristina Maggio Ombretta Viapiana Jurgen Sota Carla Gaggiano Roberto Giacomelli Ludovico Luca Sicignano Raffaele Manna Alessandra Renieri Alessandra Renieri Caterina Lo Rizzo Bruno Frediani Donato Rigante Donato Rigante Luca Cantarini |
| author_facet | Antonio Vitale Laura Obici Marco Cattalini Giuseppe Lopalco Giampaolo Merlini Nicola Ricco Alessandra Soriano Francesco La Torre Elena Verrecchia Antonella Insalaco Lorenzo Dagna Masen Abdel Jaber Davide Montin Giacomo Emmi Luisa Ciarcia Sara Barneschi Paola Parronchi Piero Ruscitti Maria Cristina Maggio Ombretta Viapiana Jurgen Sota Carla Gaggiano Roberto Giacomelli Ludovico Luca Sicignano Raffaele Manna Alessandra Renieri Alessandra Renieri Caterina Lo Rizzo Bruno Frediani Donato Rigante Donato Rigante Luca Cantarini |
| author_sort | Antonio Vitale |
| collection | DOAJ |
| description | Objective: To describe the role of biotechnological therapies in patients with tumor necrosis factor receptor associated periodic syndrome (TRAPS) and to identify any predictor of complete response.Methods: Clinical, laboratory, and therapeutic data from 44 Caucasian TRAPS patients treated with biologic agents were retrospectively collected in 16 Italian tertiary Centers.Results: A total of 55 biological courses with anakinra (n = 26), canakinumab (n = 16), anti-TNF-α agents (n = 10), and tocilizumab (n = 3) were analyzed. A complete response was observed in 41 (74.5%) cases, a partial response in 9 (16.4%) cases and a treatment failure in 5 (9.1%) cases. The frequency of TRAPS exacerbations was 458.2 flare/100 patients-year during the 12 months prior to the start of biologic treatment and 65.7 flare/100 patients-years during the first 12 months of therapy (p < 0.0001). The median duration of attacks was 5.00 (IQR = 10.50) days at the start of biologics and 1.00 (IQR = 0.00) days at the 12-month assessment (p < 0.0001). Likewise, a significant reduction was observed in the Autoinflammatory Disease Activity Index during the study period (p < 0.0001). A significant corticosteroid sparing effect was observed as early as the first 12 months of treatment both in the number of patients requiring corticosteroids (p = 0.025) and in the dosages employed (p < 0.0001). A significant reduction was identified in the erythrocyte sedimentation rate (p < 0.0001), C reactive protein (p < 0.0001), serum amyloid A (p < 0.0001), and in the 24-h proteinuria dosage during follow-up (p = 0.001). A relapsing-remitting disease course (OR = 0.027, C.I. 0.001–0.841, p = 0.040) and the frequency of relapses at the start of biologics (OR = 0.363, C.I. 0.301–0.953, p = 0.034) were significantly associated with a complete response. No serious adverse events were observed.Conclusions: Treatment with biologic agents is highly effective in controlling clinical and laboratory TRAPS manifestations. Patients with a relapsing-remitting course and a lower frequency of flares at the start of treatment show more likely a complete response to biologic agents. |
| first_indexed | 2024-12-13T19:35:17Z |
| format | Article |
| id | doaj.art-5c0a3951700142319be2e2e7c72002f8 |
| institution | Directory Open Access Journal |
| issn | 2296-858X |
| language | English |
| last_indexed | 2024-12-13T19:35:17Z |
| publishDate | 2021-07-01 |
| publisher | Frontiers Media S.A. |
| record_format | Article |
| series | Frontiers in Medicine |
| spelling | doaj.art-5c0a3951700142319be2e2e7c72002f82022-12-21T23:33:50ZengFrontiers Media S.A.Frontiers in Medicine2296-858X2021-07-01810.3389/fmed.2021.668173668173Biotechnological Agents for Patients With Tumor Necrosis Factor Receptor Associated Periodic Syndrome—Therapeutic Outcome and Predictors of Response: Real-Life Data From the AIDA NetworkAntonio Vitale0Laura Obici1Marco Cattalini2Giuseppe Lopalco3Giampaolo Merlini4Nicola Ricco5Alessandra Soriano6Francesco La Torre7Elena Verrecchia8Antonella Insalaco9Lorenzo Dagna10Masen Abdel Jaber11Davide Montin12Giacomo Emmi13Luisa Ciarcia14Sara Barneschi15Paola Parronchi16Piero Ruscitti17Maria Cristina Maggio18Ombretta Viapiana19Jurgen Sota20Carla Gaggiano21Roberto Giacomelli22Ludovico Luca Sicignano23Raffaele Manna24Alessandra Renieri25Alessandra Renieri26Caterina Lo Rizzo27Bruno Frediani28Donato Rigante29Donato Rigante30Luca Cantarini31Department of Medical Sciences, Surgery and Neurosciences, Research Center of Systemic Autoinflammatory Diseases and Behçet's Disease and Rheumatology-Ophthalmology Collaborative Uveitis Center, University of Siena, Siena, ItalyAmyloidosis Research and Treatment Center, Fondazione Istituto di Ricovero e Cura a Carattere Scientifico (IRCCS) Policlinico San Matteo, Pavia, ItalyPediatric Clinic, University of Brescia and Spedali Civili di Brescia, Brescia, ItalyRheumatology Unit, Department of Emergency and Organ Transplantation, University of Bari, Bari, ItalyAmyloidosis Research and Treatment Center, Fondazione Istituto di Ricovero e Cura a Carattere Scientifico (IRCCS) Policlinico San Matteo, Pavia, ItalyDepartment of Medical Sciences, Surgery and Neurosciences, Research Center of Systemic Autoinflammatory Diseases and Behçet's Disease and Rheumatology-Ophthalmology Collaborative Uveitis Center, University of Siena, Siena, ItalyDepartment of Internal Medicine, Arcispedale Santa Maria Nuova-Istituto di Ricovero e Cura a Carattere Scientifico (IRCCS), Reggio Emilia, ItalyClinical Pediatrics, University of Bari, Bari, ItalyPeriodic Fever Research Center, Institute of Internal Medicine, Catholic University of the Sacred Heart, Fondazione Policlinico A. Gemelli, Rome, ItalyDivision of Rheumatology, Department of Pediatric Medicine, Bambino Gesù Children's Hospital, Istituto di Ricovero e Cura a Carattere Scientifico (IRCCS), Rome, ItalyUnit of Immunology, Rheumatology, Allergy and Rare Diseases, Istituto di Ricovero e Cura a Carattere Scientifico (IRCCS) San Raffaele Hospital, Vita-Salute San Raffaele University, Milan, Italy0Rheumatology Unit, Santa Chiara Hospital, Trento, Italy1Division of Immunology and Rheumatology, Department of Paediatric Infectious Diseases, University of Turin, Regina Margherita Children's Hospital, Turin, Italy2Department of Experimental and Clinical Medicine, University of Florence, Florence, ItalyDepartment of Medical Sciences, Surgery and Neurosciences, Research Center of Systemic Autoinflammatory Diseases and Behçet's Disease and Rheumatology-Ophthalmology Collaborative Uveitis Center, University of Siena, Siena, ItalyDepartment of Medical Sciences, Surgery and Neurosciences, Research Center of Systemic Autoinflammatory Diseases and Behçet's Disease and Rheumatology-Ophthalmology Collaborative Uveitis Center, University of Siena, Siena, Italy2Department of Experimental and Clinical Medicine, University of Florence, Florence, Italy3Division of Rheumatology, Department of Biotechnological and Applied Clinical Science, University of L'Aquila, L'Aquila, Italy4Department of Health Promotion Sciences Maternal and Infantile Care, Internal Medicine and Medical Specialties “G. D'Alessandro”, University of Palermo, Palermo, Italy5Rheumatology Section, Department of Medicine, University of Verona, Verona, ItalyDepartment of Medical Sciences, Surgery and Neurosciences, Research Center of Systemic Autoinflammatory Diseases and Behçet's Disease and Rheumatology-Ophthalmology Collaborative Uveitis Center, University of Siena, Siena, Italy6Clinical Pediatrics, Department of Molecular Medicine and Development, University of Siena, Siena, Italy7Department of Medicine, Università Campus Bio-Medico di Roma, Rome, ItalyPeriodic Fever Research Center, Institute of Internal Medicine, Catholic University of the Sacred Heart, Fondazione Policlinico A. Gemelli, Rome, ItalyPeriodic Fever Research Center, Institute of Internal Medicine, Catholic University of the Sacred Heart, Fondazione Policlinico A. Gemelli, Rome, Italy8Medical Genetics, University of Siena, Siena, Italy9Genetica Medica, Azienda Ospedaliera Universitaria Senese, Siena, Italy8Medical Genetics, University of Siena, Siena, Italy0Rheumatology Unit, Department of Medical Sciences, University Hospital of Siena (Azienda Ospedaliera Universitaria Senese, AOUS), Siena, Italy1Department of Life Sciences and Public Health, Fondazione Policlinico A. Gemelli IRCCS, Rome, Italy2Università Cattolica Sacro Cuore, Rome, ItalyDepartment of Medical Sciences, Surgery and Neurosciences, Research Center of Systemic Autoinflammatory Diseases and Behçet's Disease and Rheumatology-Ophthalmology Collaborative Uveitis Center, University of Siena, Siena, ItalyObjective: To describe the role of biotechnological therapies in patients with tumor necrosis factor receptor associated periodic syndrome (TRAPS) and to identify any predictor of complete response.Methods: Clinical, laboratory, and therapeutic data from 44 Caucasian TRAPS patients treated with biologic agents were retrospectively collected in 16 Italian tertiary Centers.Results: A total of 55 biological courses with anakinra (n = 26), canakinumab (n = 16), anti-TNF-α agents (n = 10), and tocilizumab (n = 3) were analyzed. A complete response was observed in 41 (74.5%) cases, a partial response in 9 (16.4%) cases and a treatment failure in 5 (9.1%) cases. The frequency of TRAPS exacerbations was 458.2 flare/100 patients-year during the 12 months prior to the start of biologic treatment and 65.7 flare/100 patients-years during the first 12 months of therapy (p < 0.0001). The median duration of attacks was 5.00 (IQR = 10.50) days at the start of biologics and 1.00 (IQR = 0.00) days at the 12-month assessment (p < 0.0001). Likewise, a significant reduction was observed in the Autoinflammatory Disease Activity Index during the study period (p < 0.0001). A significant corticosteroid sparing effect was observed as early as the first 12 months of treatment both in the number of patients requiring corticosteroids (p = 0.025) and in the dosages employed (p < 0.0001). A significant reduction was identified in the erythrocyte sedimentation rate (p < 0.0001), C reactive protein (p < 0.0001), serum amyloid A (p < 0.0001), and in the 24-h proteinuria dosage during follow-up (p = 0.001). A relapsing-remitting disease course (OR = 0.027, C.I. 0.001–0.841, p = 0.040) and the frequency of relapses at the start of biologics (OR = 0.363, C.I. 0.301–0.953, p = 0.034) were significantly associated with a complete response. No serious adverse events were observed.Conclusions: Treatment with biologic agents is highly effective in controlling clinical and laboratory TRAPS manifestations. Patients with a relapsing-remitting course and a lower frequency of flares at the start of treatment show more likely a complete response to biologic agents.https://www.frontiersin.org/articles/10.3389/fmed.2021.668173/fulltumor necrosis factor receptor-associated periodic syndromebiologic therapypersonalized medicineinterleukin-1 inhibitorstumor necrosis factor inhibitorstocilizumab |
| spellingShingle | Antonio Vitale Laura Obici Marco Cattalini Giuseppe Lopalco Giampaolo Merlini Nicola Ricco Alessandra Soriano Francesco La Torre Elena Verrecchia Antonella Insalaco Lorenzo Dagna Masen Abdel Jaber Davide Montin Giacomo Emmi Luisa Ciarcia Sara Barneschi Paola Parronchi Piero Ruscitti Maria Cristina Maggio Ombretta Viapiana Jurgen Sota Carla Gaggiano Roberto Giacomelli Ludovico Luca Sicignano Raffaele Manna Alessandra Renieri Alessandra Renieri Caterina Lo Rizzo Bruno Frediani Donato Rigante Donato Rigante Luca Cantarini Biotechnological Agents for Patients With Tumor Necrosis Factor Receptor Associated Periodic Syndrome—Therapeutic Outcome and Predictors of Response: Real-Life Data From the AIDA Network Frontiers in Medicine tumor necrosis factor receptor-associated periodic syndrome biologic therapy personalized medicine interleukin-1 inhibitors tumor necrosis factor inhibitors tocilizumab |
| title | Biotechnological Agents for Patients With Tumor Necrosis Factor Receptor Associated Periodic Syndrome—Therapeutic Outcome and Predictors of Response: Real-Life Data From the AIDA Network |
| title_full | Biotechnological Agents for Patients With Tumor Necrosis Factor Receptor Associated Periodic Syndrome—Therapeutic Outcome and Predictors of Response: Real-Life Data From the AIDA Network |
| title_fullStr | Biotechnological Agents for Patients With Tumor Necrosis Factor Receptor Associated Periodic Syndrome—Therapeutic Outcome and Predictors of Response: Real-Life Data From the AIDA Network |
| title_full_unstemmed | Biotechnological Agents for Patients With Tumor Necrosis Factor Receptor Associated Periodic Syndrome—Therapeutic Outcome and Predictors of Response: Real-Life Data From the AIDA Network |
| title_short | Biotechnological Agents for Patients With Tumor Necrosis Factor Receptor Associated Periodic Syndrome—Therapeutic Outcome and Predictors of Response: Real-Life Data From the AIDA Network |
| title_sort | biotechnological agents for patients with tumor necrosis factor receptor associated periodic syndrome therapeutic outcome and predictors of response real life data from the aida network |
| topic | tumor necrosis factor receptor-associated periodic syndrome biologic therapy personalized medicine interleukin-1 inhibitors tumor necrosis factor inhibitors tocilizumab |
| url | https://www.frontiersin.org/articles/10.3389/fmed.2021.668173/full |
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