Neuroretinitis is an atypical presentation of ocular leptospirosis. Patients present with neuroretinitis as the only sign of leptospirosis which poses a diagnostic challenge. We reported two cases of ocular leptospirosis with neuroretinitis presentation in healthy individuals. Both patients presented swollen discs, macula star and retinal vasculitis. None of the patients had systemic symptoms of leptospirosis before the ocular complaint. Both patients were treated with oral doxycycline for four weeks and recovered well.
