Efficacy of Tocilizumab in Conventional Treatment-Refractory Adult-Onset Still's Disease
Por:
Ortiz-Sanjuan, F, Blanco, R, Calvo-Rio, V, Narvaez, J, Romero, ER, Olive, A, Castaneda, S, Flores, AG, Hernandez, MV, Mata, C, Vilamajo, IR, Giraldo, WAS, Caracuel, MA, Freire, M, Arango, CG, Llobet, J, Arija, SM, Marras, C, Moll-Tuduri, C, Plasencia-Rodriguez, C, Rosello, R, Urruticoechea, A, Velloso-Feijoo, ML, del Blanco, J, Gonzalez-Vela, MC, Rueda-Gotor, J, Pina, T, Loricera, J, Gonzalez-Gay, MA
Publicada:
1 jun 2014
Resumen:
Objective. Adult-onset Still's disease (AOSD) is frequently refractory to standard therapy. Tocilizumab (TCZ) has demonstrated efficacy in single cases and in small series of patients with AOSD. The aim of this multicenter study was to assess the efficacy of TCZ in patients with AOSD refractory to conventional treatment.
Methods. This was a retrospective open-label study of TCZ treatment in 34 patients with AOSD who had experienced an inadequate response to corticosteroids and at least 1 standard synthetic immunosuppressive drug and also, in many cases, biologic agents.
Results. The mean +/- SD age of the patients (8 men and 26 women) was 38.7 +/- 16.1 years. The median duration of AOSD before TCZ was initiated was 4.2 years (interquartile range [IQR] 1-9 years). The initial dosages of intravenous TCZ were 8 mg/kg every 4 weeks in 22 patients, 4 mg/kg every 4 weeks in 2 patients, and 8 mg/kg every 2 weeks in 10 patients. TCZ treatment resulted in rapid and maintained improvement in both clinical and laboratory parameters. After 1 year of TCZ therapy, the incidence of joint manifestations had decreased from 97.1% at baseline to 32.4%, the incidence of both cutaneous manifestations and fever had decreased from 58.8% to 5.9%, and the incidence of lymphadenopathy had decreased from 29.4% to 0%. A dramatic reduction in laboratory markers of inflammation, including the C-reactive protein level, the erythrocyte sedimentation rate, and the ferritin level, was achieved. The median dosage of prednisone was also reduced, from 13.8 mg/day (IQR 5-45) at the initiation of TCZ to 2.5 mg/day (IQR 0-30) at 12 months. After a median followup of 19 months (IQR 12-31 months), only 2 patients required permanent discontinuation of TCZ therapy because of severe infections.
Conclusion. TCZ treatment was associated with rapid and maintained clinical and laboratory improvement in patients with AOSD refractory to standard treatment. However, joint manifestations seem to be more refractory to treatment compared with systemic manifestations.
Filiaciones:
Ortiz-Sanjuan, F:
Hosp Univ Marques de Valdecilla, Santander 39008, Spain
Blanco, R:
Hosp Univ Marques de Valdecilla, Santander 39008, Spain
Calvo-Rio, V:
Hosp Univ Marques de Valdecilla, Santander 39008, Spain
Narvaez, J:
Hosp Univ Bellvitge, Barcelona, Spain
Romero, ER:
Hosp Univ Virgen del Rocio, Seville, Spain
Olive, A:
Hosp Badalona Germans Trias & Pujol, Badalona, Spain
Castaneda, S:
IIS Princesa Madrid, Hosp Univ La Princesa, Madrid, Spain
Flores, AG:
Hosp Merida, Merida, Spain
Hernandez, MV:
Hosp Clin Barcelona, Barcelona, Spain
Mata, C:
Hosp Univ Marques de Valdecilla, Santander 39008, Spain
Vilamajo, IR:
Hosp Son Llatzer, Palma De Mallorca, Spain
Giraldo, WAS:
Hosp Ramon & Cajal, E-28034 Madrid, Spain
Caracuel, MA:
Hosp Reina Sofia, Cordoba, Spain
Freire, M:
Hosp Univ Juan Canalejo, La Coruna, Spain
Arango, CG:
Hosp Univ Basurto, Bilbao, Spain
Llobet, J:
Hosp Santa Creu & Sant Pau, Barcelona, Spain
Arija, SM:
Urgencias Hosp Reg Univ Carlos Haya, Malaga, Spain
Marras, C:
Hosp Univ Virgen de la Arrixaca, Murcia, Spain
Moll-Tuduri, C:
Hosp Mateu Orfila, Mahon, Spain
Plasencia-Rodriguez, C:
Hosp Univ La Paz, Madrid, Spain
Rosello, R:
Hosp San Jorge, Huesca, Spain
Urruticoechea, A:
Hosp Can Misses, Ibiza, Spain
Velloso-Feijoo, ML:
Hosp Valme, Seville, Spain
del Blanco, J:
Hosp St Jaume, Calella, Spain
Gonzalez-Vela, MC:
Hosp Univ Marques de Valdecilla, Santander 39008, Spain
Rueda-Gotor, J:
Hosp Univ Marques de Valdecilla, Santander 39008, Spain
Pina, T:
Hosp Univ Marques de Valdecilla, Santander 39008, Spain
Loricera, J:
Hosp Univ Marques de Valdecilla, Santander 39008, Spain
Gonzalez-Gay, MA:
Hosp Univ Marques de Valdecilla, Santander 39008, Spain
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