A keratitis with an unusual, sessile, filamentary mass extending into the anterior chamber developed in a patient three weeks after penetrating keratoplasty. The causative organism was identified as Exophilia (Wangiella) dermatitidis, a dematiaceous fungus. The infection was cured with a combination of medical and surgical therapy. Inoculation of the isolate into rabbit corneas produced a similar keratitis from which the same organism was cultured. Miconazole levels measured in corneal tissue removed at surgery were approximately 25 times greater than the minimum inhibitory concentration for the fungal isolate.