Peripheral ulcerative keratitis (PUK) is an aggressive, potentially sight-threatening cause for peripheral corneal thinning. It is thought to be the result of immune complex deposition at the limbus, resulting in corneal inflammation and stromal melt. We present a case of a 43-year-old female patient of African origin, presenting with PUK and associated corneal perforation as the primary presentation of HIV infection. An urgent tectonic deep anterior lamellar keratoplasty was performed under general anaesthesia with excellent outcome. The patient was referred to the sexual health clinic and anti-retroviral treatment was initiated. This case is to the best of our knowledge the first report from the UK of PUK with corneal perforation as the primary presentation of HIV infection. As highlighted in this report, infection with HIV may initially be silent; therefore, it is vital to consider HIV infection when dealing with PUK of unknown aetiology.