Purpose: A 40-year-old male with a severe corneal melting, peripheral infiltration and nasal perforation in the right eye after a not affiliated case of urethritis is reported. Methods: Retrospective case report. Results: A sample of conjunctival discharge was sent to culture, in where Gram-diplococci grew and therefore a systemic antibiotic treatment was initiated. Given the severity of the clinic, a penetrating keratoplasty is conducted two days after admission and the corneal button was sent to microbiology, which polymerase chain reaction was positive for Neisseria gonorrhoeae. Conclusion: Despite it being a rare process in an adult, keratitis caused by Neisseria gonorrhoeae should be suspected in all sexually active patients with conjunctivitis and with a clinic genitourinary medical history. The systemic therapy showed a superior effectiveness in combination with topical therapy than the last one on its own.