Dario Roccatello1, Savino Sciascia, Daniela Rossi, Mirella Alpa, Carla Naretto, Alessandra Russo, Elisa Menegatti, Simone Baldovino. 1. Dipartimento di Malattie Rare, Immunologiche, Ematologiche ed Immunoematologiche, Centro di Ricerche di Immunopatologia e Documentazione su Malattie Rare (CMID), Struttura Complessa a Direzione Universitaria di Immunologia Clinica, Ospedale Torino Nord Emergenza San G. Bosco ed Università di Torino, Italia. dario.roccatello @ unito.it
Abstract
BACKGROUND: Current therapies have changed systemic vasculitis from a disease with a high rate of mortality to a chronic curable condition. A limited percentage of patients either remains refractory to conventional treatment or experiences dose-limiting side effects. METHODS: 11 patients (4 affected by idiopathic systemic microscopic polyangiitis, 5 by Wegener's granulomatosis, and 2 by Churg-Strauss syndrome) intolerant or refractory to conventional therapies including cyclophosphamide were enrolled. All patients received rituximab as a rescue therapy and were followed for 30-54 months. Following rituximab administration, immunosuppressive drugs were rapidly tapered and no immunosuppressive maintenance therapy was given. RESULTS: Significant decreases in levels of serum creatinine, proteinuria, erythrocyte sedimentation rate, C-reactive protein, and ANCA titers were observed during the follow-up (at least 30 months after rituximab administration). Arthralgia and weakness rapidly disappeared in all patients. Out of 7 patients, 5 reported a decrease in the degree of paresthesia and in the electrophysiologic parameters. Six months after rituximab administration the mean dose of prednisone was 5.5 mg/day. CONCLUSION: In this sample of patients with systemic vasculitis who were refractory or intolerant to more conventional treatment, rituximab proved to be safe and effective in a long-term follow-up, and showed steroid- and immunosuppressive-sparing effects allowing the persistence of long-lasting remissions without maintenance therapy.
BACKGROUND: Current therapies have changed systemic vasculitis from a disease with a high rate of mortality to a chronic curable condition. A limited percentage of patients either remains refractory to conventional treatment or experiences dose-limiting side effects. METHODS: 11 patients (4 affected by idiopathic systemic microscopic polyangiitis, 5 by Wegener's granulomatosis, and 2 by Churg-Strauss syndrome) intolerant or refractory to conventional therapies including cyclophosphamide were enrolled. All patients received rituximab as a rescue therapy and were followed for 30-54 months. Following rituximab administration, immunosuppressive drugs were rapidly tapered and no immunosuppressive maintenance therapy was given. RESULTS: Significant decreases in levels of serum creatinine, proteinuria, erythrocyte sedimentation rate, C-reactive protein, and ANCA titers were observed during the follow-up (at least 30 months after rituximab administration). Arthralgia and weakness rapidly disappeared in all patients. Out of 7 patients, 5 reported a decrease in the degree of paresthesia and in the electrophysiologic parameters. Six months after rituximab administration the mean dose of prednisone was 5.5 mg/day. CONCLUSION: In this sample of patients with systemic vasculitis who were refractory or intolerant to more conventional treatment, rituximab proved to be safe and effective in a long-term follow-up, and showed steroid- and immunosuppressive-sparing effects allowing the persistence of long-lasting remissions without maintenance therapy.
Authors: Z Chocova; Z Hruskova; H Mareckova; B Svobodova; D Duskova; V Bednarova; E Jancova; R Rysava; V Tesar Journal: Clin Rheumatol Date: 2014-11-12 Impact factor: 2.980