| Autor: |
Mikael Ebbo, Aurélie Grados, Maxime Samson, Matthieu Groh, Anderson Loundou, Aude Rigolet, Benjamin Terrier, Constance Guillaud, Clarisse Carra-Dallière, Frédéric Renou, Agnieszka Pozdzik, Pierre Labauge, Sylvain Palat, Jean-Marie Berthelot, Jean-Loup Pennaforte, Alain Wynckel, Céline Lebas, Noémie Le Gouellec, Thomas Quémeneur, Karine Dahan, Franck Carbonnel, Gaëlle Leroux, Antoinette Perlat, Alexis Mathian, Patrice Cacoub, Eric Hachulla, Nathalie Costedoat-Chalumeau, Jean-Robert Harlé, Nicolas Schleinitz |
| Jazyk: |
angličtina |
| Rok vydání: |
2017 |
| Předmět: |
|
| Zdroj: |
PLoS ONE, Vol 12, Iss 9, p e0183844 (2017) |
| Druh dokumentu: |
article |
| ISSN: |
1932-6203 |
| DOI: |
10.1371/journal.pone.0183844 |
| Popis: |
To assess efficacy and safety of rituximab (RTX) as induction therapy, maintenance of remission and treatment of relapses in a cohort of IgG4-related disease (IgG4-RD) patients.Nationwide retrospective multicenter study of IgG4-RD patients treated with at least one course of RTX. Clinical, biological and radiological response, relapse rate and drug tolerance were analyzed. Kaplan-Meier curves were plotted and risk factors for relapse studied with a Cox regression model.Among 156 IgG4-RD patients included in the French database, 33 received rituximab. Clinical response was noted in 29/31 (93.5%) symptomatic patients. Glucocorticoids withdrawal was achieved in 17 (51.5%) patients. During a mean follow-up of 24.8 ±21 months, 13/31 (41.9%) responder patients relapsed after a mean delay of 19 ±11 months after RTX. Active disease, as defined by an IgG4-RD Responder Index >9 before RTX, was significantly associated with relapse (HR = 3.68, 95% CI: 1.1, 12.6) (P = 0.04), whereas maintenance therapy with systematic (i.e. before occurrence of a relapse) RTX retreatment was associated with longer relapse-free survival (41 versus 21 months; P = 0.02). Eight severe infections occurred in 4 patients during follow-up (severe infections rate of 12.1/100 patient-years) and hypogammaglobulinemia ≤5 g/l in 3 patients.RTX is effective for both induction therapy and treatment of relapses in IgG4-RD, but relapses are frequent after B-cell reconstitution. Maintenance therapy with systematic RTX infusions is associated with longer relapse-free survival and might represent a novel treatment strategy. Yet, the high rate of infections and the temporary effect of RTX might be hindrances to such strategy. |
| Databáze: |
Directory of Open Access Journals |
| Externí odkaz: |
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