AIM: To report a successful osteo-odonto keratoprosthesis(OOKP) procedure in a case of end stage of corneal blindness due to Stevens-Johnson syndrome (SJS). METHODS: An interventional case report. RESULTS: We describe a 35-year-old Indian woman, a known case of SJS with bilateral dry eyes and corneal blindness (failed corneal graft with vascularised total corneal opacity in the right eye and non-healing corneal ulcer in the left eye). Vision was hand movement only in both eyes. The corneal ulcer healed with medical treatment resulting in vascularised total corneal opacity with no improvement in vision. OOKP was performed in the right eye and the vision was improved from hand movement to 6/6. The same vision was maintained in the right eye at the last follow-up 5 years after surgery. CONCLUSION: OOKP provides good visual rehabilitation with long-term anatomically stable prosthesis in patients with end-stage of ocular surface disorders and corneal blindness secondary to SJS.