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Title: | Efficacy of Anakinra in Refractory Adult-Onset Still's Disease: Multicenter Study of 41 Patients and Literature Review. |
Author: | Ortiz Sanjuán, Francisco Blanco, Ricardo Riancho Zarrabeitia, Leyre Castañeda, Santos Olivé Marqués, Alejandro Riveros, Anne Velloso Feijoo, María L. Narváez García, Francisco Javier Jiménez Moleón, Inmaculada Maiz Alonso, Olga Ordóñez, María del Carmen Bernal, José A. Hernández, María V. Sifuentes Giraldo, Walter A. Gómez Arango, Catalina Galíndez Agirregoikoa, Eva Blanco Madrigal, Juan Ortiz Santamaria, Vera Blanco Barnusell, Jordi del Dios, Juan R. de Moreno, Mireia Fiter, Jordi Riscos, Marina de los Carreira, Patricia Rodriguez Valls, María J. González Vela, M. Carmen Calvo Río, Vanesa Loricera, Javier Palmou Fontana, Natalia Pina, Trinitario Llorca Díaz, Javier González-Gay, Miguel A. |
Keywords: | Malalties rares Reumatologia Medicaments Estudi de casos Ús terapèutic Rare diseases Rheumatology Drugs Case studies Therapeutic use |
Issue Date: | 1-Sep-2015 |
Publisher: | Lippincott, Williams & Wilkins. Wolters Kluwer Health |
Abstract: | Adult-onset Still's disease (AOSD) is often refractory to standard therapy. Anakinra (ANK), an interleukin-1 receptor antagonist, has demonstrated efficacy in single cases and small series of AOSD. We assessed the efficacy of ANK in a series of AOSD patients.Multicenter retrospective open-label study. ANK was used due to lack of efficacy to standard synthetic immunosuppressive drugs and in some cases also to at least 1 biologic agent.Forty-one patients (26women/15 men) were recruited. They had a mean age of 34.414 years and a median [interquartile range (IQR)] AOSD duration of 3.5 [2-6] years before ANK onset. At that time the most common clinical features were joint manifestations 87.8%, fever 78%, and cutaneous rash 58.5%. ANK yielded rapid and maintained clinical and laboratory improvement. After 1 year of therapy, the frequency of joint and cutaneous manifestations had decreased to 41.5% and to 7.3% respectively, fever from 78% to 14.6%, anemia from 56.1% to 9.8%, and lymphadenopathy from 26.8% to 4.9%. A dramatic improvement of laboratory parameters was also achieved. The median [IQR] prednisone dose was also reduced from 20 [11.3-47.5] mg/day at ANK onset to 5 [0-10] at 12 months. After a median [IQR] follow-up of 16 [5-50] months, the most important side effects were cutaneous manifestations (n=8), mild leukopenia (n=3), myopathy (n=1), and infections (n=5).ANK is associated with rapid and maintained clinical and laboratory improvement, even in nonresponders to other biologic agents. However, joint manifestations are more refractory than the systemic manifestations. |
Note: | Reproducció del document publicat a: https://doi.org/10.1097/MD.0000000000001554 |
It is part of: | Medicine, 2015, vol. 94, num. 39, p. e1554 |
URI: | http://hdl.handle.net/2445/127430 |
Related resource: | https://doi.org/10.1097/MD.0000000000001554 |
ISSN: | 0025-7974 |
Appears in Collections: | Articles publicats en revistes (Ciències Clíniques) Articles publicats en revistes (Institut d'lnvestigació Biomèdica de Bellvitge (IDIBELL)) |
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