AIM: To report a successful intrastromal injection of amphotericin B 5mg/L in a refractory fungal keratitis.METHODS: An interventional case report RESULTS: A 48-year-old lady presented with history of redness of the right eye for one week duration followed by decrease in vision and corneal opacity for two days. There was no histow of trauma or foreign body. Examination revealed visual acuity of 6/16 with pinhole of 6/12 of the right eye. The conjunctiva was injected with minimal eye discharge. There was a full thickness stromal abscess at the paracentral area of the cornea. It was irregular,feathery margin with few satellite lesions. There was no epithelial defect noted. Hypopyon level was also seen.The left eye was normal. A presumptive diagnosis of fungal keratitis was entertained. Despite three weeks of intensive treatment with topical amphotericin B every 2 hours and natamycin every 4 hours and antibiotic cover,the lesion showed no sign of resolution. It grew larger and a new focal lesion of stromal abscess appeared at the 12 o'clock position. We decided to proceed with an intrastromal injection of amphotericin B 5mg/L in lieu of therapeutic penetrating keratoplasty. The size of the ulcer was substantially reduced with total disappearance of hypopyon. There was no ocular toxicity observed following the intervention. Patient regained her normal visual acuity of 6/6 after 2 months of intervention.CONCLUSION: An intrastromal injection of amphotericin B 5mg/L provides an alternative method of treating refractory fungal keratitis. It is also shown to be an effective and safe procedure with promising results.