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THU0471 Tocilizumab in Refractory Adult-Onset Still’s Disease: Multicenter Study of 27 Patients
  1. F. Ortiz-Sanjuán1,
  2. R. Blanco1,
  3. F. Narváez2,
  4. E. Rubio-Romero3,
  5. S. Castañeda4,
  6. M. Hernández5,
  7. W. Sifuentes-Giraldo6,
  8. I. Ros Vilamajó7,
  9. A. Mas7,
  10. A. Gallego Flores8,
  11. S. Manrique-Arija9,
  12. C. Gómez Arango10,
  13. R. Roselló11,
  14. C. Marras12,
  15. C. Plasencia-Rodriguez13,
  16. J. Llobet14,
  17. M. Velloso-Feijoo15,
  18. M. Freire16,
  19. M. Caracuel17,
  20. M. Moll Tudurí18,
  21. P. Lluch18,
  22. J. Loricera1,
  23. V. Calvo-Río1,
  24. M. González-Gay1
  1. 1Valdecilla, Santander
  2. 2Hospital Bellvitge, Barcelona
  3. 3HU Virgen del Rocío, Sevilla
  4. 4H Princesa, Madrid
  5. 5H Clínic, Barcelona
  6. 6H Ramón y Cajal, Madrid
  7. 7H Son Llàtzer, Palma de Mallorca
  8. 8H, Mérida
  9. 9HRU Carlos Haya, Málaga
  10. 10HU Basurto, Bilbao
  11. 11H San Jorge, Huesca
  12. 12HUVA Arrixaca, Murcia
  13. 13HU La Paz, Madrid
  14. 14H Sant Pau, Barcelona
  15. 15H Valme, Sevilla
  16. 16HUCA, La Coruña
  17. 17H Reina Sofía, Córdoba
  18. 18H Mateu Orfila, Mahón, Spain

Abstract

Background Adult-onset Still’s disease (AOSD) is often refractory to standard immunosuppressive therapy. Tocilizumab (TCZ) has shown efficacy in isolated cases or in small series.

Objectives We assess the efficacy of TCZ in AOSD.

Methods Multicenter study of 27 patients with AOSD of 18 hospitals diagnosed according to Yamagouchi’s criteria (J Rheumatol 1992;19:424). TCZ was used due to lack of good response to standard therapy or to other biologic agents.

Results The 27 patients (20 women/ 7 men), had a mean age of 37.2±15.9 (range 16-71) and an average duration of AOSD of 5.9±4.6 years (range 0.1-17) to onset of TCZ. Prior to the onset of TCZ and besides corticosteroids, patients had recived the following drugs: Metotrexate (26 patients), Anakinra (12), Etanercept (6), Adalimumab (5) and Infliximab (3). TCZ standard dose was 8 mg/k/iv/4 weeks.

At TCZ onset, the most frequent clinical manifestations were joint (27 cases), cutaneous (14) and fever (18), along with analytical abnomalities, increase of ESR or CRP (20 cases), anemia (11) or leukocytosis (15). Clinical and analytical improvement was observed soon, 1st month after the onset of TCZ therapy (TABLE). After a mean follow-up of 20.8±12 months, cutaneous manifestations disappeared in 13 of 14 patients (92,9%), fever in 17 of 18 (94.4%) and joint manifestation in 22 of 27 (81,5%). Improvement of analytical abnormalities was observed in most cases with normalization of the blood cell count in 9 of 15 (60%) patients, anemia in 11 of 11 (100%), ESR in 15 of 19 (78.9 % ), CRP in 17 of 20 (85%), hepatic enzymes (AST/ALT) in 3 of 4 (50 %) and ferritin seric levels in 11 of 13 (84,6%). The median [IQR] dose of steroids was reduced from 15 [8.8-25] to 5 [1.3-7.5].

Conclusions In refractory AOSD, TCZ yields early and maintained clinical-analytical response, even in refractory cases to other biological agents. Although TCZ showed global efficacy, joint are more refractory than other systemic manifestations.

Disclosure of Interest None Declared

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