Purpose: To present a case of peripheral ulcerative keratitis with perforation in the setting of syphilis. Method: A single observational case report in urban USA. Case Presentation: A patient with multiple sexual partners presented with bilateral peripheral ulcerative keratitis, unilateral corneal perforation, and a pustular rash. Workup was positive for syphilis as well as HSV, HIV, and multiple autoantibody tests. Findings improved after treatment for syphilis. Conclusion: Bilateral peripheral ulcerative keratitis in syphilis has been reported in one prior case. However, corneal perforation in the setting of syphilis infection has not, to our knowledge, been reported previously.