OBJECTIVE To present a case of bilateral peripheral ulcerative keratitis (PUK) caused by primary herpes simplex virus-1 (HSV-1) infection resulting in corneal perforation. METHODS A 24-year-old man presented at the eye casualty of our clinic, with a 20-day history of severe pain, redness, photophobia, and tearing in both of his eyes. Slit-lamp examination revealed bilateral superior corneal perforation. A laboratory work-up that included immunological testing for infectious and autoimmune factors showed primary HSV infection. Positive PCR analysis of corneal scrapings for HSV confirmed initial end-organ ocular infection. Because the patient showed progressive HSV-1-related PUK resulting in bilateral superior corneal perforation with iris prolapse, he was prescribed both systemic and topical acyclovir and prednisone. He then underwent bilateral surgical intervention, namely eccentric penetrating keratoplasty in one eye and a two step procedure in the other, whereby two corneal patch grafts and an amniotic membrane transplant were initially used, followed 1 month later by a large diameter penetrating keratoplasty. CONCLUSIONS In cases of PUK, differential diagnosis should include infectious and autoimmune diseases. Primary HSV should also be considered as a potential cause of this form of keratitis, which, if left untreated, can lead to devastating outcomes. To our knowledge, this is the first published case of bilateral PUK caused by primary HSV-1 infection.