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Efficacy of Anakinra in Refractory Adult-Onset Still's Disease: Multicenter Study of 41 Patients and Literature Review

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 (26 women/15 men) were recruited. They had a mean age of 34.4 ± 14 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.

 Fuente: Medicine (Baltimore) 2015 Sep;94(39):e1554

 Año de publicación: 2015

Nº de páginas: 8

Tipo de publicación: Artículo de Revista

 DOI: 10.1097/MD.0000000000001554

ISSN: 0025-7974,1536-5964

Autores/as

ORTÍZ SANJUÁN, F

BLANCO, R

RIANCHO ZARRABEITIA, L

CASTAÑEDA, S

OLIVÉ, A

RIVEROS, A

VELLOSO FEIJOO, ML

NARVÁEZ, J

JIMÉNEZ MOLEÓN, I

MAIZ ALONSO, O

ORDOÑEZ, C

BERNAL, JA

HERNÁNDEZ, MV

SIFUENTES GIRALDO, WA

GÓMEZ ARANGO, C

GALÍNDEZ AGIRREGOIKOA, E

BLANCO MADRIGAL, J

ORTÍZ SANTAMARÍA, V

BLANCO BARNUSELL, J DEL

DIOS, JR DE

MORENO, M

FITER, J

RISCOS, M DE LOS

CARREIRA, P

RODRÍGUEZ VALLS, MJ

GONZÁLEZ VELA, MC

CALVO RIO, V

LORICERA, J

PALMOU FONTANA, N

PINA, T