Objective: To describe the findings and the imaging characteristics of a patient with poppers maculopathy. Methods: The ocular findings were evaluated with fundus autofluorescence imaging, spectral optical coherence tomography and optical coherence tomography angiography. Results: A 36-year-old man presented with a history of blurring vision after using poppers. Best-corrected visual acuity was 0.9 and intraocular pressure was normal in both eyes. Fundus examination revealed hyperreflective appearence at macula with a faint foveolar reflex. Optical coherence tomography revealed focal hyperreflectivity and irregularity of the ellipsoid zone at the fovea. Fundus autofluorescence was normal. Optical Coherence Tomography Angiography revealed no vascular changes. The patient received hyperbaric oxygen therapy for 10 days. After one month VA increased, and OCT improved. Conclusion: Inhalation of poppers may be associated with bilateral vision loss due to the disruption of photoreseptors which is clearly demonstrated with OCT.