The first case of postoperative endophthalmitis after cataract surgery caused by Aspergillus ustus, a species that has only rarely been implicated in human disease, is described. Six weeks after cataract surgery, a 67-year-old medically controlled diabetic patient presented with uveitis, mild ciliary injection and ocular discomfort. Anterior chamber paracentesis, vitreous tap and finally complete vitrectomy with removal of the capsular bag including the intraocular lens were performed and several sets of culture yielded A. ustus. Despite vigorous systemic (itraconazole and caspofungin) and intravitreal (amphotericin B and caspofungin) antifungal therapy, the endophthalmitis did not improve. The painful eye with marked inflammation was finally enucleated. In vitro susceptibility testing of the isolate showed that it appeared resistant to amphotericin B, caspofungin, itraconazole, voriconazole, and posaconazole, and susceptible to terbinafine.
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