The effectiveness of the anti-tumor necrosis factor therapy infliximab in neuro-Behçet's disease: a systematic review and meta-analysis

Abstract

Objective: To study the effectiveness of infliximab for the treatment of refractory central neuro-Behçet's disease.

Methods: In this systematic review and meta-analysis, the research question was designed using the 'Population, Intervention, Comparator, and Outcomes' (PICO) model and the search methodology was developed according to the PRISMA statement. The study was registered on PROSPERO. Web of Science, PubMed, and Cochrane Library databases were searched for articles published in English between January 2000 and January 2020. Data were analysed using Meta-Essentials software, version 10.12. Treatment effect size was determined by a random effects model. Interstudy heterogeneity was explored using I² statistics. Cumulative meta-analysis was conducted to assess the temporal trend for accumulating evidence.

Results: Twenty-one studies, comprising 64 patients (mean age, 38 .21 years and mean disease duration, 84.76 months) were included. Effect-size analysis showed that 93.7% of the treated patients in the analysis were responders to infliximab therapy (95% confidence interval 0.88, 0.993). There was no significant inter-study heterogeneity (I² = 0%). Cumulative analysis showed accumulating evidence favoring increasing effectiveness over the last 20 years.

Conclusion: Infliximab showed considerable therapeutic effectiveness in the treatment of refractory neuro-Behçet's disease.

Keywords: Neuro-Behçet’s disease; anti-tumor necrosis factor therapy; chimeric monoclonal antibody therapy; magnetic resonance imaging; refractory disease; score for response.

Figure 1.

Flow diagram of search results and study selection (PRISMA model).

Figure 2.

Forest plot summarizing meta-analysis results of the effectiveness of infliximab to treat neuro-Behçet’s disease. Overall, 94% of patients responded to infliximab. Dark squares represent study weight, with larger square size representing higher study weight (sample size). Horizontal lines depict 95% confidence intervals. Red broken vertical line represents the summary for the proportion of patients who responded to infliximab. Studies are shown with first author name and year of publication. Ev, responders (patients with response score ≥3 according to Hamdy and Woldeamanuel neuro-Behcet’s simple response score, 2020); Trt, patients who were treated with infliximab.

Figure 3.

Forest plot summarizing cumulative meta-analysis of 7 studies showing a temporal trend indicating accumulating evidence for increasing effectiveness of infliximab to treat neuro-Behçet’s disease. Dark squares represent study weight, with larger square size representing higher study weight (sample size). Horizontal lines depict 95% confidence intervals. Red broken vertical line represents the summary for the proportion of patients who responded to infliximab.

Figure 4.

Funnel plot and standardized residual histogram for publication bias.