Identifier to cite or link to this item: http://hdl.handle.net/20.500.13003/17371
Efficacy of Anakinra in Refractory Adult-Onset Still's Disease Multicenter Study of 41 Patients and Literature Review
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AuthorOrtiz-Sanjuan, Francisco; Blanco, Ricardo; Riancho-Zarrabeitia, Leyre; Castaneda, Santos; Olive, Alejandro; Riveros, Anne; Velloso-Feijoo, Maria L.; Narvaez, Javier; Jimenez-Moleon, Inmaculada; Maiz-Alonso, Olga; Ordonez, Carmen; Bernal, Jose A.; Hernandez, Maria V.; Sifuentes-Giraldo, Walter A.; Gomez-Arango, Catalina; Galindez-Agirregoikoa, Eva; Blanco-Madrigal, Juan; Ortiz-Santamaria, Vera; del Blanco-Barnusell, Jordi; De Dios, Juan R.; Moreno, Mireia; Fiter, Jordi; de los Riscos, Marina; Carreira, Patricia; Rodriguez-Valls, Maria J.; Gonzalez-Vela, M. Carmen; Calvo-Rio, Vanesa; Loricera, Javier; Palmou-Fontana, Natalia; Pina, Trinitario; Llorca, Javier; Gonzalez-Gay, Miguel A.
Document typeresearch article
CitationOrtiz-Sanjuan F, Blanco R, Riancho-Zarrabeitia L, Castaneda S, Olive A, Riveros A, et al. Efficacy of Anakinra in Refractory Adult-Onset Still's Disease Multicenter Study of 41 Patients and Literature Review. Medicine (Baltimore). 2015 Sep;94(39):e1554.
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.
Interleukin 1 Receptor Antagonist Protein
Drug Therapy, Combination
Persona de Mediana Edad
Proteína Antagonista del Receptor de Interleucina 1