Efficacy of etanercept in the tumor necrosis factor receptor-associated periodic syndrome: a prospective, open-label, dose-escalation study
Ariel C Bulua, Douglas B Mogul, Ivona Aksentijevich, Harjot Singh, David Y He, Larry R Muenz, Michael M Ward, Cheryl H Yarboro, Daniel L Kastner, Richard M Siegel, Keith M Hull, Ariel C Bulua, Douglas B Mogul, Ivona Aksentijevich, Harjot Singh, David Y He, Larry R Muenz, Michael M Ward, Cheryl H Yarboro, Daniel L Kastner, Richard M Siegel, Keith M Hull
Abstract
Objective: To investigate the efficacy of etanercept in improving the symptoms and underlying inflammation in patients with tumor necrosis factor receptor-associated periodic syndrome (TRAPS).
Methods: Fifteen patients with TRAPS were enrolled in a prospective, open-label, dose-escalation study. Patients recorded attacks, symptom severity, and use of ancillary medications in a daily diary. Blood samples were collected during each period and measured for levels of acute-phase reactants. Between 7 years and 9 years after the conclusion of the initial study, patients completed a followup survey and were evaluated to determine the long-term outcome of etanercept treatment.
Results: Etanercept treatment significantly attenuated the total symptom score and reduced the frequency of symptoms. Etanercept also reduced levels of acute-phase reactants, particularly during asymptomatic periods. During a 10-year followup period, patients continued to receive etanercept for a median of 3.3 years, with a number of patients switching to anti-interleukin-1β receptor therapy or not receiving biologic agents, most frequently citing injection site reactions and lack of efficacy as reasons for discontinuation. However, patients continuing to receive etanercept had reduced symptoms at followup.
Conclusion: Etanercept reduces symptoms and serum levels of inflammatory markers of TRAPS in a dose-dependent manner, but does not completely normalize symptoms or acute-phase reactant levels. Although long-term adherence to etanercept is poor, continuing to receive etanercept may provide continued symptomatic benefit.
Trial registration: ClinicalTrials.gov NCT00001373.
Conflict of interest statement
The authors declare no financial conflicts of interest.
Copyright © 2012 by the American College of Rheumatology.
Figures
![Figure 1. Effect of etanercept on symptom…](https://www.ncbi.nlm.nih.gov/pmc/articles/instance/3882089/bin/nihms524767f1.jpg)
Figure 2. Reduction in serum acute phase…
Figure 2. Reduction in serum acute phase reactants with etanercept therapy in TRAPS
CRP, SAA,…
Figure 3. Biologic therapy decreases attack severity…
Figure 3. Biologic therapy decreases attack severity in the short- and long-term
Maximum and average…
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- Clinical Trial
- Research Support, N.I.H., Extramural
- Research Support, Non-U.S. Gov't
- Dose-Response Relationship, Drug
- Drug Substitution
- Etanercept
- Familial Mediterranean Fever / drug therapy*
- Humans
- Immunoglobulin G / therapeutic use*
- Immunologic Factors / therapeutic use*
- Prospective Studies
- Receptors, Tumor Necrosis Factor / therapeutic use*
- Receptors, Tumor Necrosis Factor, Type I
- Remission Induction
- Treatment Outcome
- Immunoglobulin G
- Immunologic Factors
- Receptors, Tumor Necrosis Factor
- Receptors, Tumor Necrosis Factor, Type I
- Etanercept
- ClinicalTrials.gov/NCT00001373
- Full Text Sources
- Other Literature Sources
- Medical
![Figure 2. Reduction in serum acute phase…](https://www.ncbi.nlm.nih.gov/pmc/articles/instance/3882089/bin/nihms524767f2.jpg)
![Figure 3. Biologic therapy decreases attack severity…](https://www.ncbi.nlm.nih.gov/pmc/articles/instance/3882089/bin/nihms524767f3.jpg)
Source: PubMed