Cytomegalovirus (CMV) has been associated with corneal endotheliitis, leading to endothelial cell loss and corneal decompensation. CMV corneal endotheliitis recurrence can affect between 5-60% of eyes after cessation of anti-CMV therapy. Each recurrence of endotheliitis has a detrimental effect on corneal endothelium with endothelial cell loss (ECL) exacerbated by each episode. This retrospective case series included 10 eyes of patients with proven CMV-related corneal endotheliitis, treated with maintenance dose of topical 0.5% ganciclovir four times per day and 1% prednisolone acetate once or twice a day. After one year of therapy four patients had mild inflammatory reaction but no corneal changes, which resolved with increase of topical steroid. Out of those four patients, one suffered recurrence of CMV-related endotheliitis on self-discontinuation of maintenance therapy for one week. The mean final ECL did not decrease compared with the baseline density in patients free of recurrences, and all 10 eyes had a clear graft or corneas at the end of the study. This small case series suggests topical therapy of ganciclovir and steroid could be an effective maintenance therapy in CMV-proved endotheliitis, although bigger studies are needed.