Abstract
Central and peripheral nervous system involvement may occur during the course of Behçet’s disease (BD). In vivo corneal confocal microscopy (CCM) can detect corneal small fiber damage and immune cell density. The aim of this study was to assess central corneal sensitivity, corneal subepithelial nerve plexus morphology and dendritic cell (DC) density in patients with BD. Forty-nine consecutive patients with BD and 30 healthy control subjects were included in this cross-sectional study conducted at a tertiary referral university hospital. Central corneal sensitivity was measured using the contact corneal esthesiometer (Cochet-Bonnet; Luneau, France). The laser scanning CCM (Heidelberg, Germany) was used to quantify corneal nerve fiber density (NFD), nerve branch density (NBD), nerve fiber length (NFL), and DC density. There was a significant reduction in NFD (P = 0.001) and NFL (P = 0.031) and an increase in DC density (P = 0.038) in patients with BD compared to healthy controls, whereas corneal sensitivity (P = 0.066) and NBD (P = 0.067) did not differ significantly. There was no difference in corneal sensitivity, corneal nerve parameters, or DC density between BD patients with [n = 18 (36.7%)] and without a previous history of uveitis (P > 0.05 for all). Disease duration [median (IQR), 6.5 (4.0–14.5) years] correlated with corneal sensitivity (ρ = −0.463; P = 0.001) and NFD (ρ = −0.304; P = 0.034) and corneal sensitivity correlated with NFD (ρ = 0.411; P = 0.003) and NFL (ρ = 0.295; P = 0.039) in patients with BD. CCM demonstrates corneal sub-basal nerve fiber loss and increased DC density, providing a non-invasive ophthalmic means to identify peripheral neuropathy and inflammation in patients with BD.
Highlights
Behçet’s disease (BD) is a chronic relapsing vascular-inflammatory disease, typically characterized by oro-genital ulcers, ocular inflammation and cutaneous manifestations
nerve fiber density (NFD) (P = 0.001) and nerve fiber length (NFL) (P = 0.031) were significantly reduced and dendritic cell (DC) density was increased (P = 0.038) in patients with BD compared to controls (Table 1; Figure 3)
There was no significant difference in central corneal sensitivity (P = 0.066) and nerve branch density (NBD) (P = 0.067) in BD patients compared to controls
Summary
Behçet’s disease (BD) is a chronic relapsing vascular-inflammatory disease, typically characterized by oro-genital ulcers, ocular inflammation and cutaneous manifestations. While brain injury is well documented with parenchymal and vascular involvement [7], there are studies showing spinal cord and peripheral nerve participation [5, 8,9,10]. An axonal neuropathy has been demonstrated in nerve biopsy and electrophysiological studies in patients with BD [9, 11] and a peripheral neuropathy has been described in children with BD [12]. In a study of 26 patients with BD without neuropathic symptoms, there was electrophysiological evidence of an axonal neuropathy [13]. A detailed electrophysiological study of 63 patients with BD showed abnormal nerve conduction and F-wave latencies in 14% [8]. A study of 111 patients showed that 19.8% of patients with BD had neuropathic pain, suggestive of underlying small fiber pathology [18]
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