We report a rare, potentially sight-threatening ocular complication due to central venous stenosis related to a previous site of haemodialysis catheter. A dialysis-dependent woman in her 60s presented with left eye redness for 1 month followed by a remarkably prominent vessel on the left upper eyelid for 2 weeks. Examinations found left eyelid oedema with prominent venous dilatation on the upper eyelid as well as left eye mild proptosis, conjunctiva injection with corkscrew vessels, raised intraocular pressure, and dilated and tortuous retina vessels. Central thoracic venogram showed total occlusion on the left brachiocephalic vein with retrograde reflux to the jugular vein. An endovascular percutaneous transluminal balloon angioplasty was performed for the left brachiocephalic vein stenosis, which resolved the orbital, facial and neck venous congestion. The patient remained asymptomatic after 1 year.