To report a case of recurrent fungal sclerokeratitis and endophthalmitis with a very successful outcome due to aggressive combined surgical and medical therapy. To discuss the management of this potentially devastating infection.A 65-year-old man presented with 6 months of left eye redness and irritation after injury from organic matter propelled from an airboat. Initially, he had been treated with foreign body removal, antibiotics, and steroids. He was diagnosed with reactive sclerokeratitis at presentation and was treated with steroids. However, when he did not improve, cultures were obtained and Acremonium species filamentous fungi was identified. Despite treatment with appropriate topical and systemic antifungals, his fungal sclerokeratitis progressed to endophthalmitis. Two therapeutic penetrating keratoplasties (PKs) with iridectomy and intraocular amphotericin B were necessary to eradicate the fungal infection.Visual acuity was restored to 20/25-3 with correction 9 months after initial presentation. There was no recurrence of fungal infection after the second therapeutic PK.The possible reasons for recurrence of fungal infection are discussed. The role of timely and aggressive medical and surgical intervention for fungal sclerokeratitis and endophthalmitis in restoring excellent vision is emphasized.