Oxford Eye Hospital, Oxford University Hospitals NHS Foundation Trust, Oxford OX3 9DU, UK.
Nuffield Laboratory of Ophthalmology, Nuffield Department of Clinical Neurosciences, University of Oxford, Oxford OX3 9DU, UK.
Genes (Basel). 2024 Sep 8;15(9):1178. doi: 10.3390/genes15091178.
Wagner syndrome is a rare autosomal dominant vitreoretinopathy caused by mutations in chondroitin sulphate proteoglycan 2 (CSPG2)/Versican (VCAN). Here, we present a retrospective case series of a family pedigree with genetically confirmed Wagner syndrome (heterozygous VCAN exon 8 deletion), as follows: a 34-year-old mother (P1), 12-year-old daughter (P2), and a 2-year-old son (P3). The phenotype included early-onset cataract (P1), optically empty vitreous with avascular membranes (P1, 2), nasal dragging of optic nerve heads associated with foveal hypoplasia (P1, 2), tractional retinoschisis on optical coherence tomography (P2), and peripheral circumferential vitreo-retinal interface abnormality resembling white-without-pressure (P3) progressing to pigmented chorio-retinal atrophy (P1, 2). P2 developed a macula-off retinal detachment, which was treated initially with encircling band + vitrectomy + gas, followed by vitrectomy + heavy silicone oil tamponade for re-detachment from new inferior breaks. Strong vitreo-retinal adhesion was noted intraoperatively, which prevented the separation of posterior hyaloid beyond the equator. Electroretinograms from P1&2 demonstrated attenuated b-waves, a-waves, and flicker responses in light- and dark-adapted conditions, suggestive of generalised retinal dysfunction. Our patients demonstrated the clinical spectrum of Wagner syndrome, highlighting nasal dragging with foveal disruption as a distinguishing feature from other inherited vitreoretinopathies. Surgical outcomes demonstrate significant challenges in managing vitreo-retinal traction and need for further research into strategies to prevent sight loss.
瓦格纳综合征是一种罕见的常染色体显性遗传性玻璃体视网膜病变,由软骨素硫酸盐蛋白聚糖 2(CSPG2)/神经节苷脂(VCAN)基因突变引起。在此,我们报告一个经基因证实的瓦格纳综合征(杂合性 VCAN 外显子 8 缺失)家系的回顾性病例系列:一位 34 岁的母亲(P1)、12 岁的女儿(P2)和 2 岁的儿子(P3)。表型包括早发性白内障(P1)、玻璃体光学空伴无血管膜(P1、2)、视神经头鼻侧牵拉伴黄斑发育不良(P1、2)、光学相干断层扫描显示牵拉性视网膜劈裂(P2)和周边环形玻璃体视网膜界面异常(P3),类似于白色无压力(P3),进展为色素性脉络膜视网膜萎缩(P1、2)。P2 发生黄斑裂孔性视网膜脱离,最初采用环扎带+玻璃体切除术+气体治疗,随后因新的下方裂孔复发性脱离,改为玻璃体切除术+重硅油填充。术中发现玻璃体视网膜强烈粘连,阻碍了后玻璃体赤道部的分离。P1 和 P2 的视网膜电图显示明适应和暗适应时 b 波、a 波和闪烁反应减弱,提示广泛的视网膜功能障碍。我们的患者表现出瓦格纳综合征的临床谱,强调了鼻侧牵拉伴黄斑破坏是其区别于其他遗传性玻璃体视网膜病变的特征。手术结果表明,管理玻璃体视网膜牵引存在显著挑战,需要进一步研究预防视力丧失的策略。