Purpose: To report the use of topical valganciclovir for the treatment of hypertensive anterior uveitis associated with clinical signs of cytomegalovirus (CMV) iritis. Methods: A case report and review of the literature. Results: A 37-year-old man was referred with a unilateral hypertensive anterior uveitis with keratic precipitates suggestive of CMV as the causative agent. After institution of oral valganciclovir and topical corticosteroids, the patient's ocular inflammation resolved and intraocular pressure normalized. Therapy was eventually changed from oral valganciclovir to ophthalmic 1% valganciclovir ointment, which was able to effectively control ocular inflammation and allow the patient to discontinue topical corticosteroids and antihypertensive medications. Topical application of valganciclovir did not result in clinically evident ocular surface toxicity. Conclusions: 1% valganciclovir ointment may prove to be an effective treatment of hypertensive anterior uveitis associated with clinical signs of CMV iritis.