Remission of Behcet's disease with anti-tumor necrosis factor monoclonal antibody therapy: a case report

Abstract

Background: Behcet's disease (BD) is a chronic relapsing multisystem inflammatory disorder with mucocutaneous, ocular, articular, vascular, gastrointestinal and central nervous system manifestations. Tumor necrosis factor (TNF)-alpha is believed to play a pivotal role in BD. Therapeutic blockade of the activity of TNF has been successfully given in a short course of therapy with favorable effects in patients with BD refractory to conventional immunosuppressive drugs. We aimed to find out whether a 12-month treatment with infliximab, a chimeric monoclonal antibody to TNF-alpha, had any beneficial effect in reducing relapses of a patient with long-standing BD refractory to conventional immunosuppressive drugs.

Update on the therapy of Behçet disease

“…In a similar manner, infliximab was shown to be effective in reducing the various mucocutaneous manifestations of the disease [Chan and Lee, 2012;Aikawa et al 2011;Kasugai et al 2010;Lestre et al 2009;Olivieri et al 2008;Connolly et al 2005;Takayama et al 2013;Gulli et al 2003]. …”

“…In uveoretinitis, a large number of case reports/ series [Munoz-Fernandez et al 2001;Sfikakis et al 2001;Capella and Foster, 2012;Alokaily et al 2010;Olivieri et al 2008;Takamoto et al 2007;Merino et al 2006;Lindstedt et al 2005;Lanthier et al 2005;Wechsler et al 2004;Sayarlioglu et al 2004a;Rosenbaum, 2004;Giansanti et al 2004;Falappone et al 2004;Morris et al 2003;Gulli et al 2003;Triolo et al 2002;Takayama et al 2013], open-label clinical trials Handa et al 2011;Giardina et al 2011;Tanaka et al 2010;Adan et al 2010;Al-Rayes et al 2008;Tognon et al 2007;Niccoli et al 2007;Accorinti et al 2007;Tugal-Tutkun et al 2005;Benitez-del-Castillo et al 2005;Arayssi et al 2005;Sfikakis et al 2004;Ohno et al 2004;Cantini et al 2012;Keino et al 2011], and nonrandomized comparative studies Yamada et al 2010;Markomichelakis et al 2011] showed that intravenous infliximab, as single therapy, is effective in inducing prompt suppression of ocular inflammation, decreasing the frequency of uveitis attacks and allowing gradual tapering of corticosteroids (CS). As would be expected, vision of eyes with permanent lesions due to irreversible retinal damage does not improve.…”

Infliximabe no tratamento de uveíte posterior refratária em paciente com doença de Behçet

“…Até o início de 2002, quando se cogitou usar infliximabe no paciente estudado neste caso, haviam sido descritos na literatura apenas 12 casos semelhantes tratados com infliximabe, ainda sem um consenso com relação à dose, ao número e ao intervalo das infusões. Desde 2002 diversos trabalhos têm sido realizados, alguns com número maior de pacientes, mostrando uma tendência à boa resposta com o uso do infliximabe (9)(10)(11)(12)(15)(16)(17)(18)(19)(20) , havendo apenas descrição de três casos com resposta inadequada (13,14) . O maior estudo, realizado na Grécia e publicado em março de 2004, envolveu 25 pacientes com doença de Behçet e uveíte posterior, que receberam infusão única de infliximab 5 mg/ kg, com controle da inflamação ocular já no primeiro dia pós-tratamento em 24 dos 25 pacientes.…”

Late response to anti-TNF-α therapy in refractory mucocutaneous lesions of Behçet’s disease

“…Of note, anti-TNF-a therapy may be useful in refractory severe BD, particularly in cases with ocular [3], central nervous system [4] and gastrointestinal involvement [5]. The use of this family of biological drugs has also been described in patients with recalcitrant mucocutaneous lesions, and the remarkable early and dramatic response has been associated with relatively long-duration remission [5][6][7][8][9][10][11].…”

“…The use of this family of biological drugs has also been described in patients with recalcitrant mucocutaneous lesions, and the remarkable early and dramatic response has been associated with relatively long-duration remission [5][6][7][8][9][10][11].…”