A 37-year-old woman presented to our hospital with a history of headaches, blurred vision and declining visual acuity. She had a history of obstructive sleep apnoea associated with Treacher Collins syndrome, a rare congenital disorder characterised by craniofacial deformities and obstruction of the upper airways. On funduscopy we noted bilateral papilloedema and retinal haemorrhages. A CT scan of the brain excluded a space occupying lesion and contrast-enhanced CT did not show evidence of venous sinus thrombosis. The cerebrospinal fluid opening pressure on lumbar puncture was normal. We made a diagnosis of visual loss and papilloedema secondary to chronic CO2 retention from obstructive sleep apnoea. Her vision and headaches improved remarkably following a permanent tracheostomy. This case highlights the association between obstructive sleep apnoea and papilloedema and shows that the associated visual loss can ultimately be restored with a tracheostomy.