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E-mail address: rblanco humv. E-mail address: miguelaggay hotmail. Use the link below to share a full-text version of this article with your friends and colleagues. Learn more. 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 The median dosage of prednisone was also reduced, from After a median followup of 19 months IQR 12—31 months , only 2 patients required permanent discontinuation of TCZ therapy because of severe infections.
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. Other features are sore throat or pharyngitis, myalgia, lymphadenopathy, hepatosplenomegaly, and serositis.
The pathogenesis of AOSD remains unknown. Several studies have suggested a genetic predisposition, while others have identified different infectious agents as potential triggers of the disease. These findings may explain the intermittent course of AOSD and the clinical and laboratory features that resemble those of genetic autoinflammatory syndromes. However, the efficacy of these drugs in the control of disease activity is variable, and in some cases, they are associated with potential severe side effects.
However, in most cases, information related to this issue is based on small series or case reports. Therefore, the aim of the current study was to evaluate the efficacy of TCZ in a large series of patients with AOSD refractory to other treatments. All patients had previously received standard synthetic immunosuppressive drugs and in some cases other biologic agents.
AOSD was diagnosed at the rheumatology units of 21 Spanish referral centers according to the criteria described by Yamaguchi et al Other diseases were excluded. Before the initiation of TCZ treatment, the presence of infections, including hepatitis B or hepatitis C virus infections, was excluded using clinical examination and serologic tests. Data registered by each investigator were reviewed according to a protocol agreed upon beforehand that included the collection of relevant information on the patients.