A 75-year-old man, diagnosed with primary bilateral lipoid keratopathy, who reported progressive loss of visual acuity (VA). Ophthalmological examination showed VA of 20/60 in the right eye (OD) and hand movement in the left eye (OI). Biomicroscopy showed a yellowish-white corneal opacity in the full thickness of the stroma, incomplete annular shape, respecting the visual axis in the right eye and affecting the central cornea in the left eye. Superficial and deep neovascularisation extending to the centre of the cornea was also observed. Serum lipid levels were normal. The patient underwent surgery and underwent penetrating keratoplasty with extracapsular cataract extraction and intraocular lens implantation in the OI. Postoperative treatment included topical steroids and antibiotics: 4 times/day (Tobradex®, Alcon-Cusí, Barcelona, Spain), topical cyclosporine 2%: 4 times/day and oral prednisone: 50mg/day, in a descending pattern for 2 weeks. At 1 month, biomicroscopy revealed deep corneal neovascularisation extending over the bed and graft-host interface at 3 and 6 hours, with a whitish opacity around the vessels. After obtaining informed consent for compassionate use medication, topical treatment with bevacizumab (25mg/ml) was started: 4 times/day/2 months, in a descending pattern for 6 months. At 8 months, the patient was maintained on topical bevacizumab: 1x/day. Fig. 1C shows partial regression of corneal neovascularisation with clear graft. His VA was 20/40.