Pseudallescheria boydii (P. boydii) is an uncommon ocular pathogen which previously has been identified in only 10 of 905 fungal isolates identified by the Sid Richardson Microbiology Laboratory at the Cullen Eye Institute of Baylor College of Medicine. Furthermore, only one case of postoperative P. boydii endophthalmitis and four cases of endogenous P. boydii endophthalmitis have been reported. Three of the four patients with endogenous endophthalmitis died within 4 weeks of diagnosis. We describe a second case of postoperative endophthalmitis due to this fungus. The infection was successfully eradicated following vitrectomy, corneoscleral resection, and patch graft, in addition to intraocular, topical, and oral antifungal medication. Although in vitro sensitivities are variable, P. boydii is known to be relatively resistant to amphotericin B. This points to the importance of proper cultures and sensitivities when treating cases of suspected fungal endophthalmitis. Unfortunately, the patient's eye became phthisical 6 months following the initial intervention.