Autoantibodies Against Trisulfated Heparin Disaccharide and Fibroblast Growth Factor Receptor-3 May Play a Role in the Pathogenesis of Neuropathic Corneal Pain
Autoantibodies Against Trisulfated Heparin Disaccharide and Fibroblast Growth Factor Receptor-3 May Play a Role in the Pathogenesis of Neuropathic Corneal Pain
复制标题
DOI：
10.1097/ico.0000000000003142
复制
发表时间：
2023-07-01
期刊：
影响因子：
2.8
通讯作者：
Hamrah, Pedram
中科院分区：
文献类型：
--
作者：
Bayraktutar, Betul N.;Atocha, Vanessa;Hamrah, Pedram
目的：本研究的目的是描述与假定免疫障碍性小纤维神经病（SFN）相关的神经性角膜疼痛（NCP）患者的病例，这些患者存在抗三硫酸肝素二糖（TS-HDS）或成纤维细胞生长因子受体-3（FGFR-3）的自身抗体。方法：本研究是3例NCP患者的病例系列，这些患者具有阳性抗TS-HDS和/或抗FGFR-3自身抗体和系统性SFN，并通过阳性皮肤活检结果证实。结果：所有3例患者均为女性，平均年龄为34.3 ± 6.1岁。他们患有中度至重度持续性慢性眼部不适（视觉模拟量表分别为10/10、10/10和9/10）。虽然1例患者仅发生眼部疼痛和眼恐怖症，但其他2例患者发生了额外的非眼部疼痛。其中1例患者的面部和头部疼痛，1例患者报告颈部和下背部疼痛。两名患者具有高抗TS-HDS IgM滴度，而1名患者具有高抗TS-HDS IgM和抗FGFR-3 IgG滴度。皮肤活检证实存在SFN在所有患者表现为减少表皮内神经纤维density.Conclusions：抗TS-HDS和抗FGFR-3自身抗体的存在下，与NCP患者的皮肤活检结果阳性SFN突出的潜在作用，免疫功能障碍SFN在这种疾病的发病机制。
Purpose:The aim of this study was to describe cases of patients with presumable dysimmune small-fiber neuropathy (SFN)-related neuropathic corneal pain (NCP), presenting with autoantibodies against trisulfated heparin disaccharide (TS-HDS) or fibroblast growth factor receptor-3 (FGFR-3).Methods:This study was a case series of 3 patients with NCP with positive anti-TS-HDS and/or anti-FGFR-3 autoantibodies and systemic SFN as confirmed by positive skin biopsy results.Results:All 3 patients were women with a mean age of 34.3 +/- 6.1 years. They suffered from moderate to severe persistent chronic ocular discomfort (10/10, 10/10, and 9/10 on a visual analogue scale, respectively). Although 1 patient suffered from ocular pain and photophobia alone, the other 2 patients experienced additional non-ocular pain. One of the patients had pain on her face and head, and 1 patient reported neck and lower back pain. Two patients had high anti-TS-HDS IgM titers, whereas 1 patient had both high anti-TS-HDS IgM and anti-FGFR-3 IgG titers. Skin biopsy confirmed the presence of SFN in all patients by demonstrating decreased intraepidermal nerve fiber density.Conclusions:The presence of anti-TS-HDS and anti-FGFR-3 autoantibodies in patients with NCP with positive skin biopsy findings for SFN highlights the potential role of dysimmune SFN in the pathogenesis of this disease.