Abstract
Monkeypox-related ocular disease (MPXROD) is an uncommon but potentially sight-threatening complication of monkeypox virus (MPXV) infection. While most cases improve with supportive care, some patients may develop ocular involvement. We describe a 28-year-old immunocompetent man who experienced decreased vision and paracentral dendritic corneal ulcers during the acute phase of infection. Initial treatment, directed at presumed herpetic keratitis, successfully resolved the ulcers but left a residual stromal leukoma. Subsequent inflammatory flares led to an anterior chamber paracentesis, which confirmed MPXV as the causative agent. Since first-line antiviral options were not available, therapy was adjusted to oral valaciclovir alongside tapering corticosteroids. Recurrent episodes of keratouveitis were effectively managed with repeated courses of antivirals, corticosteroids, and ocular lubricants, as well as the addition of compounded topical insulin. This case highlights that MPXROD can lead to recurrent keratouveitis even in otherwise healthy individuals. When first-line antivirals are not available, treatment must be individualized, combining antivirals, corticosteroids, supportive care, and adjunctive therapies, such as topical insulin, which can play a crucial role in preserving vision and healing corneal damage in recurrent flares.