Alban Deroux1, Chistophe Chiquet2, Laurence Bouillet3. 1. Department of Internal Medicine, University Hospital of Grenoble, CS 10217, F-38043 Grenoble Cedex 09, France; Department of Ophtalmology, University Hospital of Grenoble, F-38043, Grenoble Cedex 09, France. Electronic address: ADeroux@chu-grenoble.fr. 2. Department of Ophtalmology, University Hospital of Grenoble, F-38043, Grenoble Cedex 09, France; UJF-Grenoble 1, F-38041 Grenoble, France. 3. Department of Internal Medicine, University Hospital of Grenoble, CS 10217, F-38043 Grenoble Cedex 09, France; UJF-Grenoble 1, F-38041 Grenoble, France.
Abstract
INTRODUCTION: In Behcet's disease (BD), interleukin (IL)-6 drive the immune-mediated inflammatory process. The IL-6 receptor can be targeted using tocilizumab. As an off-label treatment, we tested its efficacy in patients with BD. METHODS: Overall, 4 patients with refractory BD were treated with tocilizumab, 8mg/kg/4 weeks. Patients were clinically and biologically assessed before administering each dose and the literature was reviewed. RESULTS: Tocilizumab was found to be safe and well tolerated. BD activity decreased significantly in all patients, and prednisone dose was reduced in all cases (up to 50% of the baseline dose). Treatment appeared effective in alleviating skin/mucosal effects, neurological involvement, and uveitis, but less effective for arthralgia and abdominal pain. A very short time lag between the onset of treatment with tocilizumab and the clinical response was observed. The literature review revealed 11 previous cases reporting improvement to BD with this treatment, and 3 previous cases without efficacy. CONCLUSIONS: We reported the most important study treating refractory BD with tocilizumab; this treatment could be safe and efficient, but will require further evaluation by randomized clinical trials.
INTRODUCTION: In Behcet's disease (BD), interleukin (IL)-6 drive the immune-mediated inflammatory process. The IL-6 receptor can be targeted using tocilizumab. As an off-label treatment, we tested its efficacy in patients with BD. METHODS: Overall, 4 patients with refractory BD were treated with tocilizumab, 8mg/kg/4 weeks. Patients were clinically and biologically assessed before administering each dose and the literature was reviewed. RESULTS:Tocilizumab was found to be safe and well tolerated. BD activity decreased significantly in all patients, and prednisone dose was reduced in all cases (up to 50% of the baseline dose). Treatment appeared effective in alleviating skin/mucosal effects, neurological involvement, and uveitis, but less effective for arthralgia and abdominal pain. A very short time lag between the onset of treatment with tocilizumab and the clinical response was observed. The literature review revealed 11 previous cases reporting improvement to BD with this treatment, and 3 previous cases without efficacy. CONCLUSIONS: We reported the most important study treating refractory BD with tocilizumab; this treatment could be safe and efficient, but will require further evaluation by randomized clinical trials.
Authors: Thomas W McNally; Erika M Damato; Philip I Murray; Alastair K Denniston; Robert J Barry Journal: Orphanet J Rare Dis Date: 2017-07-17 Impact factor: 4.123