PURPOSE: To report a case of infusion misdirection syndrome, simulating an intraoperative suprachoroidal hemorrhage, in an eye undergoing trabeculectomy for primary trabeculodysgenesis. METHODS: A 21-year-old woman underwent trabeculectomy for primary trabeculodysgenesis. Congenitally abnormal intraocular anatomy led to intraoperative and postoperative complications. RESULTS: Intraoperative anterior chamber irrigation was followed by vitreous prolapse. Eye pain, globe hardening, and shallowing of the anterior chamber were caused by infusion misdirection into the posterior segment. The eye was rapidly closed after a limited wound-site vitrectomy. Intraoperative and postoperative management led to a successful outcome. CONCLUSION: Altered intraocular anatomy set the stage for infusion misdirection syndrome, a rare but manageable complication that mimicked an expulsive suprachoroidal hemorrhage. © 2002 by Elsevier Science Inc. All rights reserved.