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The aim of this study was to evaluate the visual outcomes and graft survival rate after therapeutic keratoplasty performed for interface infection after Descemet stripping automated endothelial keratoplasty (DSAEK).This is a retrospective, interventional case series. The study population comprised 7 patients who developed unilateral post-DSAEK interface infection unresponsive to conservative treatment, with or without graft exchange, and were treated with penetrating keratoplasty (PK), 9 to 9.5 mm in diameter, with en bloc excision of the recipient cornea and DSAEK graft. The main outcome measures included best spectacle-corrected visual acuity, refractive error, histological examination, reinfection, and rejection and graft survival rates.Interface infection was diagnosed in 10 (0.92%) of 1088 eyes that underwent DSAEK at our institution between 2005 and 2013. Seven of 10 eyes (0.64% of the total) were unresponsive to conservative treatment and underwent therapeutic keratoplasty. Candida and Staphylococcus species were identified in 3 cases each, and Nocardia species was identified in 1 case. With a mean post-PK follow-up of 25.4 months (range 4–60 months), no recurrence of infection was seen in any eye, and 5 of 7 PK grafts remained clear. Best spectacle-corrected visual acuity was 20/20 in 2 eyes, better than 20/50 in 4 eyes, and 20/100 or worse in 3 eyes, in 2 of which the graft had failed within 1 year of performing the PK.Therapeutic keratoplasty is instrumental in eliminating interface infection after DSAEK, possibly leading to excellent visual outcomes with a relatively high graft survival rate.