PURPOSE: To report a patient with a history of exposure keratopathy who presented with bilateral bacterial keratitis associated with streptococcal pharyngitis and use of bilevel positive airway pressure (BiPAP). DESIGN: Case report. METHODS: Pulsed-field gel electrophoresis of patient isolates from ocular and pharyngeal cultures. RESULTS: Ocular and oropharyngeal cultures from a 24-month-old child with a history of cerebral palsy on BiPAP at night revealed the same strain of Streptococcus pyogenes. CONCLUSIONS: Use of mechanical ventilation, such as BiPAP, may precipitate inoculation of eye with respiratory or oropharyngeal pathogens. To the authors' knowledge this is the first report demonstrating the association between concurrent streptococcal pharygitis, keratitis, and use of BiPAP.