We reviewed the specimen records log of the McGee Eye Institute Histopathology Service from January 1979 to December 1990 for penetrating keratoplasty specimens submitted with a clinical diagnosis of Peters' anomaly. The records of the 19 cases thereby identified were reviewed for clinical outcome and visual rehabilitation, and the histopathology of all specimens was reviewed and correlated with the clinical diagnosis. Two specimens from the same patient were eliminated as histopathologically inconsistent with Peters' anomaly; one patient's data were not used because the patient was an adult when first grafted. Six eyes were grafted two or more times, for a total of 26 grafts on 16 eyes in 10 patients. Mean age at the time of first transplant was 18 weeks (range, 3 weeks to 40 months). Mean follow up was 30 months (range, 7 months to 6 1/2 years). Five eyes had preoperative glaucoma which persisted postoperatively. Ten eyes developed glaucoma postoperatively. Of the 15 eyes with glaucoma, 14 were uncontrolled medically and 12 underwent cyclodestructive or Molteno filtering procedures, or both, to control pressure. Graft rejection developed in 9 of the 10 eyes that required a cyclodestructive procedure, with partial or complete graft failure occurring shortly after the procedure. Glaucoma did not develop in one eye; in one other eye, it was medically controlled. These 2 eyes maintained clear grafts. Of the 6 eyes that were regrafted, only 1 obtained ambulatory vision. Of the 26 eyes receiving grafts, the grafts failed completely in 17 and partially in 5; 4 have remained clear. Five patients maintain ambulatory vision.